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Surgery
of the
TRACHEA Bronchi and
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Hermes C. Grillo, MD
Surgery
of the
TRACHEA Bronchi and
HERMES C. GRILLO, MD Professor Emeritus of Surgery Department of Surgery Harvard Medical School Senior Surgeon, Chief Emeritus of General Thoracic Surgery Department of Surgery Massachusetts General Hospital Boston, Massachusetts
Illustrated by Edith Tagrin
2004 BC Decker Inc Hamilton • London
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DEDICATION
To my teachers, my colleagues, and my students—whose roles so often coincided.
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CONTENTS
Preface . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . xi Acknowledgments . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . xiii Contributors . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . xv INTRODUCTION: Development of Tracheal Surgery: A Historical Review . . . . . . . . . . . . . . . . . . . . . . 1 PART 1 DISEASES, DIAGNOSIS, RESULTS OF TREATMENT ANATOMY, PHYSIOLOGY, PATHOLOGY, DIAGNOSTIC METHODS 1. Anatomy of the Trachea . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 39 2.
Physiology of the Trachea . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 61 David J. Kanarek
3.
Pathology of Tracheal Tumors . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 73 A. Epithelial Tumors of the Trachea . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 73 Javad Beheshti, Eugene J. Mark, Fiona Graeme-Cook B. Mesenchymal Tumors of the Trachea . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 86 Javad Beheshti, Eugene J. Mark C. Tumor-Like Lesions of the Trachea . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 98 Eugene J. Mark, Javad Beheshti
4.
Imaging the Larynx and Trachea . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 103 Jo-Anne O. Shepard, Alfred L. Weber
5.
Diagnostic Endoscopy. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 161
DISEASES AND RESULTS OF TREATMENT 6. Congenital and Acquired Tracheal Lesions in Children . . . . . . . . . . . . . . . . . . . . . . . . . . . . 173 7.
Primary Tracheal Neoplasms . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 207
8.
Secondary Tracheal Neoplasms. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 249
9.
Tracheal and Bronchial Trauma . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 271
10.
Tracheostomy: Uses, Varieties, Complications . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 291
11.
Postintubation Stenosis . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 301
12.
Acquired Tracheoesophageal and Bronchoesophageal Fistula . . . . . . . . . . . . . . . . . . . . . . . 341
13.
Tracheal Fistula to Brachiocephalic Artery . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 357
14.
Infectious, Inflammatory, Infiltrative, Idiopathic, and Miscellaneous Tracheal Lesions . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 363
15.
Tracheobronchial Malacia and Compression . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 397
16.
Bronchial Sleeve Resection . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 429 Henning A. Gaissert
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Contents
PART 2 THERAPEUTIC TECHNIQUES AND MANAGEMENT ANESTHESIA, PRE- AND POSTOPERATIVE CONSIDERATIONS AND COMPLICATIONS 17. Preoperative Considerations . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 443 18.
Anesthesia for Tracheal Surgery . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 453 Paul H. Alfille
19.
Urgent Treatment of Tracheal Obstruction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 471
20.
Postoperative Management . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 479
21.
Complications of Tracheal Reconstruction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 483
SURGICAL TECHNIQUES 22. Tracheostomy, Minitracheostomy, and Closure of Persistent Stoma . . . . . . . . . . . . . . . . . . 499 23.
Surgical Approaches . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 507
24.
Tracheal Reconstruction: Anterior Approach and Extended Resection. . . . . . . . . . . . . . . . 517
25.
Laryngotracheal Reconstruction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 549
26.
Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula . . . . . . . . . . . . . . . 569
27.
Repair of Tracheobrachiocephalic Artery Fistula . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 581
28.
Reconstruction of the Lower Trachea (Transthoracic) and Procedures for Extended Resection . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 587
29.
Carinal Reconstruction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 599
30.
Main and Lobar Bronchoplasty . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 619 Douglas J. Mathisen
31.
Repair of Tracheobronchial Trauma . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 631
32.
Surgery for Tracheomalacia, Tracheopathia Osteoplastica, Tracheal Compression, and Staged Reconstruction of the Trachea . . . . . . . . . . . . . . . . . . . 645
33.
Repair of Congenital Tracheal Lesions . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 665 A. Tracheoplasty for Congenital Tracheal Stenosis . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 665 Hermes C. Grillo B. Laryngotracheoesophageal Cleft Repair . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 675 Daniel P. Ryan
34.
Cervicomediastinal Exenteration and Mediastinal Tracheostomy . . . . . . . . . . . . . . . . . . . . 681
Contents
SPECIAL PROBLEMS AND MODES OF TREATMENT 35. Laryngologic Problems Related to Tracheal Surgery . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 693 Robert H. Lofgren 36.
Foreign Body Aspiration . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 707 Daniel P. Doody
37.
Laser Therapy for Tracheobronchial Lesions . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 719 Stanley M. Shapshay and Tulio A. Valdez
38.
Tracheal Appliances. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 735 Donna J. Wilson
39.
Tracheal T Tubes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 749
40.
Tracheal and Bronchial Stenting . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 763 Douglas E. Wood
41.
Radiation Therapy in the Management of Tracheal Cancer . . . . . . . . . . . . . . . . . . . . . . . . 791 Noah C. Choi
42.
The Omentum in Airway Surgery and Tracheal Reconstruction. . . . . . . . . . . . . . . . . . . . . . . 803 after Irradiation
43.
Postpneumonectomy Bronchopleural Fistula . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 815 Cameron D. Wright
44.
Airway Management in Lung Transplantation. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 827 John C. Wain
45.
Tracheal Replacement . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 839
Index. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 855
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PREFACE
This may seem a rather large book to devote to an anatomic structure that measures only 10 to 12 cm in length and that is often considered a passive conduit. In addition, lesions affecting the trachea are hardly common. Their very rarity, however, is a principal reason for this book. I have tried to distill here 40 years of experience, ranging over a period in which contemporary airway surgery essentially developed. I hope that surgeons or physicians facing a clinical airway problem can amplify their knowledge here. Contributors to this book are all practitioners, who write from mature experience. Part 1 presents basic information on the trachea, its diseases, diagnosis, and results of treatment. Part 2 provides a surgical manual plus descriptions of special problems and their management. The organization of the “manual” is based on the fact that surgical strategy often depends as much on the location and extent of a lesion as it does on its etiology. Edith Tagrin has worked long and hard to produce elegant drawings that are detailed and precise. An experienced thoracic surgeon who observed our tracheal surgery for half a year commented that one of the most important things he had learned was how different each case could be—often in subtle ways. Such differences influence operative decisions and strategies, and affect outcome in major ways. If the reader finds useful guidance in these pages, then the book will have met its goals. Hermes C. Grillo September 2003
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“A distinction can…be drawn between specialization in a technical field and concentration in a circ*mscribed area of learning. The difference lies in the tendency of a technical specialist to exclude all other subjects from his interest and study. The concentrator seeks to maintain an active curiosity and interest concerning all techniques that might be useful in his area of concentration, and views his work in proper perspective with science as a whole.” Edward D. Churchill, 1946
“…the relative success or failure of any surgical procedure lies in attention to what may on first thought appear to be unimportant small details.” Richard H. Sweet, 1950
xii
ACKNOWLEDGMENTS
I am thankful to a host of teachers, colleagues, students, friends — and let me not forget patients — who have participated in weaving the fabric of my 55 years of surgery at Massachusetts General Hospital (MGH). I know and regret that the following list of acknowledgments will inevitably be incomplete. Edward D. Churchill was a thoughtful teacher, guide, and friend. Richard H. Sweet provided impeccable standards in the craft of thoracic surgery. Both of these individuals, giants in their field, stimulated my interest in thoracic surgery. J. Gordon Scannell was a valued teacher, friend, and colleague. Afield from MGH, I learned much about thoracic surgery from Clifford Storey at US Naval Hospital, St. Albans, a billet which I thought of as a valued reward after a year of combat surgery with the First Marine Division in Korea in 1951. Earle W. Wilkins Jr, Douglas J. Mathisen, Ashby C. Moncure, John C. Wain, Cameron D. Wright, and more recent colleagues, all worked together to establish and develop the General Thoracic Surgical Unit at MGH. Drs. Mathisen and Wright have labored particularly assiduously in the vineyard of tracheal surgery. Paul S. Russell was supportive of this project in tracheal surgery from its beginning. W. Gerald Austen initiated the founding of a thoracic surgical unit in 1969, where this work progressed. David Crockett helped to obtain support for fundamental early laboratory efforts. Sue Robinson tied the thoracic efforts together as Unit Coordinator. I think fondly and with appreciation of the following research fellows and their productive efforts: Drs. Tsuyoshi Miura, Ellen Dignan, Masazumi Maeda, Yahiro Kotake, Joel D. Cooper, John Mulliken. As well, of many clinical fellows, to name but a few: Piero Zannini, Luciano Landa, Salvino Saita, Joo Hyun Kim. Further, of many thoracic surgical residents, some of whom continue to work with special interest in tracheal surgery, notably Douglas Wood, Richard Heitmiller, Joseph Newton, Mark Allen. This is an admittedly incomplete list. Colleagues in several disciplines have collaborated helpfully and productively over many years. These include Alexander MacMillan, Reginald Greene, Alfred Weber, Theresa McLoud, and Jo-Anne Shepard in Radiology; Eugene Mark in Pathology; so many able, patient, and innovative anesthetists (I cannot list them all) including Henrik Bendixen, Henning Pontoppidan, Bennie Geffin, John Bland, Roger Wilson, Paul Alfille, William Hurford, and Warren Zapol; Noah Choi in Radiotherapy; and Robert Lofgren and William Montgomery in Otolaryngology. I cannot begin to express my appreciation for the care given to patients by the devoted and skilled nurses in the operating rooms, respiratory intensive care unit, and surgical nursing unit of MGH. I must mention Ruth Dempsey, RN, who struggled so hard and effectively to get the original Thoracic Surgical Unit up and running, and who guided it so well for many years. One of my greatest pleasures has been to meet, exchange ideas, and to work with thoughtful and innovative thoracic surgeons around the world, all of whom share a keen interest in tracheal surgery. I mention only the foremost: F. Griffith Pearson of Toronto, Mikhail Perelman of Moscow, Henry Eschapasse of Toulouse, Louis Couraud of Bordeaux, and Masazumi Maeda of Shikoku, Japan. They all contributed generously to my thinking.
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xiv
Acknowledgments
I appreciate the valuable efforts of contributors to this book, who offer their special knowledge based on profound experience. Tracheal surgery, perhaps as much as any subdivision of surgery, crosses anatomic boundaries and conventional specialty jurisdictions. Solutions to its problems have arisen from a concentration of knowledge and techniques from several areas of specialization, rather than from a narrow technical specialization itself. This repeats a distinction made by Edward D. Churchill, who clearly saw the wisdom of general education in surgery as distinct from education in general surgery. He cautioned us to avoid “myopic” specialization. Edith Tagrin, friend and colleague for many years, has provided a wealth of illuminating, meticulous, and beautiful illustrations. She richly deserves the many awards and praises that she has received in the field of medical illustration. I am grateful for her contributions. The Photography Laboratory of MGH and that of the Department of Pathology deserve special thanks. This book would not exist without the indefatigable, unfailingly cheerful, and intelligent labors of my colleague and secretary of many years, Patricia Guerriero. The publisher, Brian C. Decker, has patiently and with quiet enthusiasm supported and encouraged this work for more years than either of us wishes to recall. My colleagues, Drs. James Allan, Morton Swartz, and Gus Vlahakes, reviewed parts of the manuscript and made valuable suggestions. Dr. Henning Gaissert translated seminal papers on tracheal surgery from earlier German literature. And I must express special thanks to two generous friends in Italy, both of the University of Naples, who over the years had provided me with Elysian retreats in which to work — the late, eminent Professor of Surgery Giuseppe Zannini, at his beloved villa, La Casupola, on Capri, the most beautiful of isles, and Professor of Architecture Camillo Gubitosi, at San Gismondo, the ancient monastery he so attractively restored on a hilltop in Montefollonico, Toscana. My wife, Sue Robinson, has been most patient and encouraging, although the last thing we need in our home is one more book. Hermes C. Grillo
CONTRIBUTORS
Paul H. Alfille, MD Department of Anesthesia Massachusetts General Hospital Department of Anesthesia and Critical Care Harvard Medical School Boston, Massachusetts
Hermes C. Grillo, MD Department of Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
Javad Beheshti, MD Department of Pathology Massachusetts General Hospital Department of Pathology Harvard Medical School Boston, Massachusetts
David J. Kanarek, MB, BCh, FCPSA Department of Pulmonary and Critical Care Medicine Massachusetts General Hospital Department of Medicine Harvard Medical School Boston, Massachusetts
Noah C. Choi, MD Department of Radiation Oncology Massachusetts General Hospital Department of Radiation Oncology Harvard Medical School Boston, Massachusetts Daniel P. Doody, MD Department of Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
Robert H. Lofgren, MD, FACS Department of Otolaryngology Massachusetts General Hospital Department of Otology and Laryngology Harvard Medical School Boston, Massachusetts Eugene J. Mark, MD Department of Pathology Massachusetts General Hospital Department of Pathology Harvard Medical School Boston, Massachusetts
Henning A. Gaissert, MD Department of Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
Douglas J. Mathisen, MD Department of Thoracic Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
Fiona Graeme-Cook, MD Department of Pathology Massachusetts General Hospital Department of Pathology Harvard Medical School Boston, Massachusetts
Daniel P. Ryan, MD Department of Pediatric Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
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Contributors
Stanley M. Shapshay, MS, FACS Department of Otolaryngology Boston Medical Center Department of Otolaryngology Boston University, School of Medicine Boston, Massachusetts
Alfred L. Weber, MD Department of Radiology Harvard Medical School Department of Radiology Massachusetts Eye and Ear Infirmary Boston, Massachusetts
Jo-Anne O. Shepard, MD Department of Radiology Massachusetts General Hospital Department of Radiology Harvard Medical School Boston, Massachusetts
Donna J. Wilson, RN, MSN, RRT Department of Medicine Memorial Sloan Kettering Cancer Center New York, New York
Tulio A. Valdez, MD Department of Otolaryngology Tufts-New England Medical Center Department of Otolaryngology Tufts University, School of Medicine Boston, Massachusetts John C. Wain, MD Department of Thoracic Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
Douglas E. Wood, MD Department of General Thoracic Surgery University of Washington Seattle, Washington Cameron D. Wright, MD Department of Surgery Massachusetts General Hospital Department of Surgery Harvard Medical School Boston, Massachusetts
INTRODUCTION
Development of Tracheal Surgery: A Historical Review Hermes C. Grillo, MD
Techniques of Tracheal Surgery Treatment of Tracheal Diseases Conclusion
Despite the antiquity of tracheostomy, tracheal surgery was the last anatomic subdivision of cardiothoracic surgery to develop. In 1950, Belsey observed, “The intrathoracic portion of the trachea is the last unpaired organ in the body to fall to the surgeon, and the successful solution of the problem of its reconstruction may mark the end of the ‘expansionist’ epoch in the development of surgery.”1 Following the introduction of intratracheal anesthesia, enormous strides were taken in pulmonary surgery in the 1930s, in esophageal surgery in the 1940s, and in cardiac surgery in the 1950s after cardiopulmonary bypass became a reality.2,3 In 1961, Richard Meade noted,“Carcinoma of the trachea is a rather rare lesion, and when it is found, it is usually found to be entirely inoperable. In rare instances, the lesion is so localized that the involved trachea can be resected, and with mobilization, the ends can be brought together. This is seldom true and one is faced with the problem of what to do after resection of the trachea.”4 The 1960s proved to be a decade when advances in tracheal surgery quickened.5 Thus, by 1990, resection rates for tracheal tumors reached 63% for squamous carcinoma, 75% for adenoid cystic carcinomas, and 90% for other tumors.6 The following is not a comprehensive review of the literature on tracheal surgery. Rather, it is a selective account of tracheal surgical development. Current references are not necessarily included unless they report progress in the fundamentals or significant evolution of the techniques. For historical reasons, I have therefore often referred to an author’s earlier paper rather than to more complete later reports. Emphasis is on the beginnings and early development of important concepts and procedures. I am certain that there are omissions in this account, for which I express regret. The review is divided into two parts. The first part traces the evolution of techniques of tracheal surgery. The second part records the acquisition of information about characteristics and treatment of specific diseases of the trachea. There is, of course, considerable overlap.
Techniques of Tracheal Surgery Tracheostomy Even a brief history must note the ancient use of tracheostomy for a variety of indications. The story has been traced by a number of authors.7–10 Although Aretaeus and Galen remarked on the use of tracheostomy in the
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Surgery of the Trachea and Bronchi
second and third centuries, the arteria aspera, the “rough artery,” as the trachea was known for generations, only slowly entered the surgical theater. The specific technique of Antyllus in the fourth century CE is recorded.7 Fabricius of Aquapendente, who introduced the idea of a tracheostomy tube, warned of the danger of this intervention. Tracheostomy was regarded with fear and considered inappropriate by most. In 1546, Antonio Musa Brasavola of Ferrara treated a pharyngeal abscess by tracheostomy after the patient had been refused by barber surgeons. In his thorough and excellent review, Goodall identified this as the first recorded successful tracheostomy, despite many ancient references to the trachea and possibly to its opening.7 Marco Aurelio Severino used tracheostomy during an epidemic of diphtheria in Napoli in 1610, performing it through a vertical incision recommended by Fabricius (Gerolamo Fabrizio d’Aquapendente).11 In Paris, 1620, Nicholas Habicot performed tracheostomies, which he termed “bronchotomy,” for one patient who had blood clots in the trachea, and for another who attempted to foil a highwayman by swallowing a bag of gold coins which then stuck in his esophagus and compressed the airway. Tracheostomy relieved the obstruction. We have no record of what happened to the bag of gold. Surprisingly contemporary tracheostomy devices are illustrated in seventeenth-century texts, including Habicot’s Question Chirurgicale (Figure 1), Sanctorius’ (Santorio Santorio) Commentaria in 1625, and Julius Casserius’ Tabulae Anatomicae in 1627. Thomas Fienus of Louvain used the word “tracheotomy” in 1649, although it was not often so called for another century. Over the centuries, a few reports of successful tracheostomies have been made.7 A drowning victim was treated with tracheostomy by Georges Détharding in 1714. In 1720, René-Jacques Croissant de Garengeot described the division of the thyroid isthmus in order to accomplish a tracheostomy, using a long vertical incision that went almost from chin to sternum. He further argued that failure of the tracheostomy was often due to its being performed too late. Lorenz Heister, in 1718, is said to have been the first to use the word “tracheostomy.” In 1730, George Martin described an inner cannula for the tracheostomy tube, a device suggested to him by a layman. Chovell, in 1732, performed a tracheostomy at the request of a patient who faced death by hanging. Unfortunately, this did not save the accused. Goodall found reports of 28 successful tracheostomies done prior to 1825, when Pierre Bretonneau in Tours used tracheostomy with success in treating “croup.”7 In Paris, 1831, Bretonneau’s pupil, Armand Trousseau, applied the technique in management of diphtheria, saving about a quarter of 200 children who were dying from the disease. Tracheostomy changed little technically, although controversy continued about its indications, locations, and hazards.9 Chevalier Jackson largely cast it in its modern form, cautioning against tracheostomy high in the trachea.12 He believed that “tracheotomy is the worst done of all operations.”13 Tracheostomy found application in general anesthesia, but was soon displaced by endotracheal intubation.14 As diphtheria waned, tracheostomy was used in poliomyelitis, to prevent infection, in head and chest injuries, after major surgery, and to reduce dead space. The endotracheal tube largely replaced tracheostomy as a preferred method to establish an emergency airway. Later, tracheostomy vied with endotracheal intubation for management of secretions, and, subsequently, as a route for mechanical positive pressure ventilation. A high incidence of complications was recognized even prior to the frequent appearance of postintubation injuries.8,15,16 Plastic surgical closure of a persistent tracheostomy by a cutaneous inversion technique was described by Lawson and Grillo in 1970.17
Repair and Healing of the Airway An ancient concern that cast a shadow on tracheal surgery into the twentieth century was that cartilage healed poorly. Hippocrates had cautioned, “The most difficult fistulae are those which occur in the cartilaginous areas….”18 In the second century CE, Aretaeus pronounced, “The lips of the wound do not coalesce, for they are both cartilaginous and not of a nature to unite.”7 As late as 1990, Naef repeated that “tracheobronchial tissue, as compared to the stomach, intestine, or even skin, does not heal well… both the rigidity and the poor blood supply of the cartilaginous structure are definitely major handicaps.”19
Development of Tracheal Surgery: A Historical Review
FIGURE 1 Tracheostomy pictured by Nicolas Habicot in Question Chirurgicale. Par laquelle il est demonstré que le chirurgien doit assurément pratiquer l’operation de la bronchotomie. J. Corrozet, Paris, l620. A, The patient. B, The larynx. C, The wound or bronchotomy. D, The instrument for bronchotomy. E, The hollow cannula. F, The straps for fastening it on the neck. G, Plain smooth band to apply over the cannula to scatter the air stream. H, Needle to suture the wound when one removes the dressing to make the wound heal.
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Nonetheless, examples of early attempts and sometimes success in bronchial and tracheal repairs after trauma are recorded. Indeed, The Rigveda, a book of Hindu medicine dating from between 2000 and 1000 BCE, noted that the trachea can reunite “when the cervical cartilages are cut across, provided they are not entirely severed.”9 Ambroise Paré described suture of tracheal lacerations in the mid-1500s in three patients, the first from a sword wound, and the latter two from knife wounds.20 The first patient survived despite a concomitant injury to the internal jugular vein. The second patient suffered division of both the trachea and esophagus and died. We do not know the outcome of the third patient. Brasavola observed recovery after a suicide attempt severed five tracheal rings.7 Eventually, cumulative clinical experience in the twentieth century established that the trachea healed firmly with suture repair after laceration or rupture.21–27 Jackson and colleagues demonstrated firm healing of experimental bronchial anastomosis in 1949.28 In 1950, Daniel and colleagues confirmed fibrous tissue repair of linear tracheal incisions in the laboratory, as did Rob and Bateman clinically in 1949.29,30 Quinby and Morse pointed out experimentally, for the first time in 1911, the importance of peribronchial tissue in bronchial closure.31 In 1942, Rienhoff and colleagues made fundamental observations that bronchial healing after pneumonectomy was accomplished by new connective tissue, which grew over the ends of the stump, rather than by mucosal healing alone.32
End-To-End Tracheal and Bronchial Anastomosis Glück and Zeller, in 1881, demonstrated healing after end-to-end tracheal anastomosis in dogs and believed the technique could be applied in man.33 Colley, in 1895, in order to avoid stenosis, tried elliptical and bayonet anastomoses in dogs after resecting five rings.34 Primary anastomosis of the cervical trachea, after limited resection for post-traumatic stenosis, followed in 1886 by Küster, apparently the first in man.35 In 1898, Bruns performed an extended lateral excision of a papillary tumor in the cervical trachea, but managed the tracheal defect by packing and with a cannula.36 Complex methods for repair of cervical tracheal defects, with skin or fascia lata, were also explored in the early twentieth century by Nowakowski in 1909 and by Levit in 1912, among others.37,38 Eiselsberg successfully performed a second resection of 1.5 cm of trachea in one patient.39 Mathey and colleagues commented in 1966, “This type of radical tracheal surgery was then forgotten for half a century.”40 The era of open thoracic surgery had arrived. By 1936, Churchill had refined the technique of lobectomy to achieve a 2.6% mortality rate.41 As interest in bronchial and tracheal surgery grew by the midtwentieth century, laboratory experiments confirmed that healing followed end-to-end anastomosis of both bronchi and trachea, although sometimes with stenosis.28–30,42–45 Bronchial repair after trauma proved the feasibility of airway reconstruction. Sanger described bronchial repair in patients during World War II.46 In 1949, Griffith resected a stricture and anastomosed the bronchus 3 months after rupture.47 Other late repairs of ruptured bronchi followed.48 Scannell first performed immediate repair of a bronchus ruptured during closed injury in 1951.49 Belcher in 1950 and Mathey and Oustrieres in 1951 reported reanastomosis of main bronchi after accidental division during surgery.50,51 Earlier cautious enlargement of bronchial stenosis by wire-supported dermal grafts were replaced by resection and reconstruction.52,53 The technique was applied to low-grade tumors and to carcinomas as sleeve lobectomy evolved.54–58 The evolution of sleeve lobectomy is described in more detail in Chapter 16,“Bronchial Sleeve Resection.” Concurrent vascular sleeve resection was also pursued by Johnston and Jones.59 Main bronchial resection without removal of lung tissue was extensively described by Newton and colleagues.60
Inhibitions to Tracheal Reconstruction With retrospective wisdom, we may ask, “What were the barriers to application of the bronchoplastic and tracheal anastomotic techniques, just noted, to clinical tracheal resection and reconstruction?” I have men-
Development of Tracheal Surgery: A Historical Review
tioned a persistent suspicion that tracheal cartilage healed poorly. A second, more insistent concern was that only a very limited segment of trachea could be removed and reanastomosis accomplished. In 1909, Nowakowski placed the limit of resection at 3 to 4 cm, from cadaver studies.37 Colley and Küster respectively reported resections of three rings and 2 to 4 cm.34,35 Rob and Bateman, on the basis of cadaver dissection, placed the limit at 2 cm.30 Belsey believed that three or four rings, about 2 cm, was the limit in man.1 Cantrell and Folse placed the limit at two rings if over 80 years of age.61 Nicks cited “one inch or more” as a limit in the cervical trachea.62 These presumed limits led to devising complex methods of cervical tracheal reconstruction with available tissue flaps and transfers, and, further, to a century-long search for a means of tracheal replacement. This search ranged over foreign material in many forms, autogenous tissue constructions, tissue and foreign material complexes, fixed or “tanned” tissues, transplantation, and, recently, tissue engineering. Success has eluded investigators to date. The story of this frustrating pursuit and the reasons for its overall failure thus far are detailed in Chapter 45, “Tracheal Replacement.” An additional difficulty for reconstruction was maintenance of safe, continuous, and stable ventilation throughout the procedure, especially for intrathoracic tracheal operations. The evolution of anesthetic techniques is discussed later. Finally, primary tumors of the trachea remained rare, as can be seen from earlier chronicles of their occurrence.63,64 Stenoses from traumatic, iatrogenic, or inflammatory causes were not seen frequently before 1960. Thus, any single thoracic surgeon was not often challenged. Each case was largely dealt with in ad hoc fashion.
Primary Resection and Reanastomosis: Initial Experiences In the mid-twentieth century, the recrudescence of interest in tracheal surgery was marked by experiments in tracheal healing and replacement, and by renewed clinical efforts. Earliest attempts at reconstruction of the cervical trachea were still most often by staged, complex repairs, typified by Crafoord and Eindgren’s cutaneous reconstruction after tumor removal in 1945.65 Belsey appears to have been the first to have dared to remove intrathoracic tracheal tumors, but his repair was with wire-supported fascia, leaving a residual strip of mucosa for continuity and for epithelial regeneration.1 Clagett and colleagues and others followed, using polyethylene tubes or patches to repair the defects.66 The story of these efforts to replace the trachea partially or circumferentially is related in Chapter 45, “Tracheal Replacement.” Despite continued concerns about the feasible length of tracheal resection and lingering doubts about cartilaginous healing, a number of successful resections and reconstructions with primary anastomosis were described in the 1950s and early 1960s, most often for shorter, benign lesions such as stricture.66 Conley successfully resected the second and third rings for scar in 1953, with end-to-end anastomosis.67 Kay removed four rings of proximal trachea for leiomyoma, without event, in 1951.68 Sweet, in 1952, resected a cervical “cylindroma” with end-to-end anastomosis and questioned whether this might be possible intrathoracically.69 Macmanus and McCormick, in 1954, excised a three-ring segment for the same tumor, which lay about 2 cm above the carina, with end-to-end repair.70 An anastomotic leak was patched with fascia lata and a protective tracheostomy added. Forster and colleagues reported in 1957 and 1958 a series of three successful cervical and cervicomediastinal tracheal resections with primary anastomosis of 1.5, 4, and 3 cm for tumor, post-traumatic stenosis, and postintubation stenosis, respectively.71,72 Other similar resections were reported separately by Binet and Miscall and their colleagues.73,74 In 1959, Flavell had successfully corrected a postintubation stricture at the thoracic outlet by resection, but did this from a difficult, transthoracic approach—an error that was to be repeated later by other surgeons.75 Mattes performed a 4 cm transthoracic lower tracheal resection for cylindroma in 1958, wrapping the anastomosis with pleura.76 Indicative of the revived interest in tracheal surgery was the extensive report in 1960 by Baumann and Forster of worldwide experiences in tracheal surgery.77 They pointed out that improvements in diagnosis (endoscopy) and technical and ventilatory methods had served to widen the field beyond tra-
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cheostomy and endoscopic treatment alone. At the same time, the potential for surgery of the thoracic trachea was of exciting interest.
Anatomic Mobilization of Trachea These mid-century experiences in tracheal reconstruction, chiefly in the upper trachea, and most often of limited extent, made it clear that the basic techniques of tracheal anastomosis could achieve sound healing. The “2 cm rule,” which had served to inhibit advances in tracheal surgery, was now challenged by experimental studies reinvestigating the extent of trachea that could be removed, and approximation achieved by anatomic tracheal mobilization, without use of prosthetic replacement. Clinical experiences, especially with intrathoracic and carinal lesions, contributed to widening the possibilities for more extended resection. Ferguson and colleagues determined the extensibility of human trachea from cadavers to be 35% at 29 years and 17% at 76 years, with the most stretch reached with 200 g of tension.43 In living dogs, the majority of resectable length was obtained at 450 g of tension at the anastomosis, which is about 30 to 35% of the tracheal length. Michelson and colleagues, in an effort to increase the length of resectable trachea, freed the right main bronchus in dogs by incising the right pulmonary ligament and resecting the left main bronchus at the carina, and then reanastomosing it to the bronchus intermedius.78 This permitted resection of twelve rings in the dog. They found that the human trachea could be stretched 4 to 6 cm by mobilization, and that an added 2.5 to 5 cm could be obtained by the maneuver described in dogs. Tracheal elongation in fresh human cadavers, with the same dissection and 450 g of upward pull, allowed 2.5 to 3 cm elevation after division of the pulmonary ligament, and 5 to 6 cm after freeing the left main bronchus in four cadaver subjects under 50 years of age. Respectively, 1 to 1.5 and 2 to 3 cm were measured in four subjects at 50 to 75 years of age. Cantrell and Folse sought to determine the limits of feasibility of primary anastomosis in repair of circumferential defects.61 In resection of 20 to 58% of dog trachea, the suture line tension ranged from 400 to 2,750 g. The tension required for anastomosis varied markedly between flexed and extended neck positions. Disruption of anastomosis occurred between 1,700 and 3,100 g, at resection lengths of 46 to 63% of the trachea. However, they noted in human trachea obtained at autopsy that resection of more than 2 cm over the age of 80 produced unacceptable anastomotic tension, based on experimentally derived standards. In 1959, Harris showed radiologically that neck extension elongated the trachea by 2.6 cm.79 Som and Klein extended the length of human cadaver trachea by only 1.6 cm by circumferential incision of the intercartilaginous annular ligaments.80 Grillo and colleagues reported in 1964, from autopsy studies in man, that over half of the adult trachea could be resected and continuity reestablished by full mobilization of limiting structures (Figure 2).81 Steps in mobilization were 1) right hilar dissection and division of right pulmonary ligament, 2) division of left main bronchus, and 3) freeing pulmonary vessels from the pericardium. With the subject’s neck in neutral position, these steps permitted tracheal excisions averaging 3 cm (3 to 8 rings), 2.7 cm (3 to 12 rings), and 0.9 cm (0.5 to 3 rings), for a total of 6.4 cm (11 to 18 rings). Anastomotic tension rose exponentially with resection of successive 1 cm segments, from 25 g at 1 cm to 675 g at 7 cm. Age did not prove to be seriously limiting. This was considerably below the biologically dangerous limit of 1,700 g determined by Cantrell and Folse.61 Division of the left main bronchus allowed the advancement of the distal tracheal stump and right main bronchus. In addition, if an even more extended resection was to be necessary, division of the cervical trachea two to three rings below the cricoid allowed this segment of cervical trachea to be devolved into the mediastinum with intact lateral vascular supply.81 This maneuver proposed to allow reconstruction of the intrathoracic trachea by simple anastomosis, while permitting later staged reconstruction of the cervical trachea, which would be more safely possible. Because of the complexity of this last approach, division and reimplantation of the left main bronchus was later applied clinically, only in the case of carinal reconstruction, and then, rarely.
Development of Tracheal Surgery: A Historical Review
2 Hermes C. Grillo, MD, in the 1960s, when work on tracheal surgery was underway at Massachusetts General Hospital (MGH). He became the first Chief of General Thoracic Surgery at MGH when a specialized unit was founded in 1969. FIGURE
Stimulated by Grillo’s clinical experiences with cervical tracheal resection for postintubation stenosis, Mulliken and Grillo reported in 1968 an investigation of the amount of trachea that might be resected by cervical and mediastinal mobilization and still permit anastomosis, leaving the pleural cavity intact.82 Pretracheal mobilization was done down to the carina, with division of the thyroid isthmus, in cadavers. With the neck in 15 to 35 degrees of flexion, 1,000 to 1,200 g of tension applied to the divided tracheal ends
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permitted an average resection and reapproximation of 4.5 cm (7.2 rings). Right hilar mobilization with the pleura open allowed an increment of resection of 1.4 cm (2.5 rings), giving a total of 5.9 cm. The average tracheal length was 11 cm. Cervical flexion permitted a gain of 1.3 cm (2.5 rings) over the neutral position. Thus, cervical flexion and pretracheal mobilization alone appeared to allow significant cervical or cervicomediastinal resection and anastomosis, especially important for the postintubation lesions, which were increasing in frequency, and that often occurred in patients who could not tolerate thoracotomy. Appreciation of the possible degree of tracheal mobilization, based upon anatomic principles(ie, pretracheal mobilization, cervical flexion, hilar dissection, including intrapericardial freeing, and mobility of detached main bronchi), made possible a systematic and aggressive approach to tracheal resection and reconstruction not previously conceived. The episodic ad hoc approach, which produced single case reports, at times almost expressing a surgeon’s surprise at what he was able to accomplish, yielded to more confident and planned approaches. Using such principles, significant series of resections and reconstructions of cervical and thoracic tracheae for stenosis and tumor were reported by Grillo, Deverall, Perelman, Naef, Couraud, Pearson, Dor, Levasseur, and Harley and their colleagues.83–93
Laryngeal Release An additional dividend for extended upper tracheal resection came from otolaryngology. Ogura and colleagues had suggested dividing hyoid muscles to help close the gap produced by resection of a subglottic laryngeal stenosis.94,95 In 1969, Dedo and Fishman offered thyrohyoid laryngeal release as a necessary adjunct to tracheal resection for stenosis.96 Division of the thyrohyoid muscles, the superior cornua of the thyroid cartilage, and of the thyrohyoid membrane, with care to preserve superior laryngeal nerves, allowed the larynx to drop about 2.5 cm. Montgomery described an alternative method for laryngeal release—suprahyoid release.97 Muscle attachments to the superior surface of the hyoid bone, the stylohyoid muscles, and the hyoid bone anterior to the digastric slings were all divided, allowing the larynx to drop. It was the opinion of both Grillo and Pearson (unpublished, c. 1979) that less severe deglutitional disorders of shorter duration followed suprahyoid release than thyrohyoid release. Release is not routinely necessary for upper tracheal resection.98 Grillo observed clinically that laryngeal release did not transfer effective relaxation for lower tracheal or carinal resections.99 Valesky and colleagues confirmed this in cadaver studies.100
Tracheal Blood Supply The detailed arterial supply of the trachea was described as a necessary corollary to tracheal surgery. Grillo emphasized the entry of small segmental arteries via “lateral pedicles” of tissue attached to either side of the trachea.84 Miura and Grillo precisely defined the blood supply to the upper trachea in 1966, usually from three principal branches of the inferior thyroid artery, with the first (or lowest) branch most often predominant.101 Salassa and colleagues completed a definitive study of the tracheal blood supply in 1977, confirming Miura and Grillo’s description of the cervical tracheal supply, and mapping the arterial supply of the thoracic trachea from bronchial, supreme intercostal, subclavian, right internal thoracic, and innominate arteries.102 Segmental tracheoesophageal arteries connected often to lateral longitudinal arteries and then to transverse intercartilaginous arteries. Three to 7 tracheal arteries were found in the “lateral pedicles.” Unlike the intramural collateral of tracheal blood supply in the dog, which maintains tracheal viability despite complete circumferential dissection, and subsequent division and anastomosis, the same procedure in man has led to necrosis.61
Carinal Resection and Reconstruction At the lower end of the trachea, the special problems (anatomic, technical, and anesthesiologic) of carinal reconstruction loomed. Lesions, most often neoplastic, were centered in the carina, extended to the
Development of Tracheal Surgery: A Historical Review
carina from low in the trachea, or to the carina from main bronchi or lungs. Experimentally, Grindlay and colleagues resected right lung and carina in dogs in 1949, with end-to-end anastomosis of trachea to left main bronchus.103 Ferguson and colleagues also performed right and left pneumonectomies in dogs in 1950, with resection of carina and end-to-end anastomosis.43 In 1951, Juvenelle and Citret, working at the University of Buffalo, showed experimentally the feasibility of lateral implantation of bronchus into trachea, without loss of blood supply and without interference in ventilation.104 They further described experiments in which they resected the carina with a three to four ring segment of trachea, and then anastomosed the trachea to the right or left main bronchus and implanted the other main bronchus into the side of the trachea. They found it necessary to free the trachea to reduce otherwise excessive tension. Additionally, they remarked that freeing the trachea permitted anastomosis of the trachea directly to right and left main bronchi without excessive tension, after short segment resection.105 Meyer and colleagues experimentally implanted the right upper lobe and right main bronchus into the trachea in 1951.106 Ehrlich and colleagues, in 1952, transposed a right main bronchus to the lateral tracheal wall, and later resected the left lung and carina in dogs.107 Kiriluk and Merendino, in 1953, described a variety of experimental tracheal, bronchial, and carinal reconstructions, including reapproximation of both main bronchi to the carina and tracheobronchial anastomosis after carinal pneumonectomy.45 Nicks similarly reconstructed the carina after resection in pigs in 1956, but under hypothermia.62 In 1958, Björk and Rodriguez described experiments in reconstruction by direct anastomosis after resection of the carina and twelve tracheal rings in dogs.108 The right main bronchus was sutured end-to-end to the trachea and the left main bronchus end-to-side to the intermediate bronchus. This followed the similarly successful clinical procedure by Barclay and colleagues, described below.109 The same anastomoses after carinal resection were performed in dogs in 1969 in confirmatory studies.110 Clinically, Abbott repaired large oval defects created by right pneumonectomy and right carinal lateral excision for bronchogenic carcinoma in 5 patients in 1950, by transverse closure.111 Two of the patients died. Other patching techniques were used to repair such lateral defects, including dermal grafts, synthetic materials, and patches or flaps of retained bronchial wall.112,113 These complex and frequently unsuccessful patch techniques are reviewed in Chapter 45, “Tracheal Replacement.” In 1951, Mathey locally resected a “cylindroma” of the back wall of the trachea at the carina, including posterior walls of both proximal and main bronchi.114 Repair was effected by longitudinal suture of the medial bronchial margins and transverse suture of the remaining defect. In these years, surgeons struggled with the problem of tracheobronchial anastomosis at the carina. In 1954, Crafoord and colleagues reported anastomosis of the bronchus intermedius to the trachea at the site of main bronchial origin, after upper lobectomy and bronchial excision.115 The next year, Björk obtained access to the carina from the left chest, mobilizing the aorta after division of four pairs of intercostal arteries, in order to successfully resect the left main bronchus and anastomose its lobar bifurcation to the prior origin of the bronchus at the trachea.116 In 1959, he presented follow-up of 16 patients who had undergone bronchotracheal anastomosis, with four stenoses.117 Abbey-Smith and Nigan described a similar left-sided approach in 1979, for amputation of the left main bronchus at the carina, for pneumonectomy in a case of proximal lung tumor.118 Barclay and colleagues, in 1957, resected about 5 cm of trachea and carina to remove a recurrent adenoid cystic carcinoma.109 Division of the pulmonary ligament allowed anastomosis of the trachea to the right main bronchus. The left main bronchus was anastomosed end-to-side to the bronchus intermedius. Intermittent ventilation sufficed for the second anastomosis. A second patient was handled identically. Both patients recovered. The authors reported in the same paper that dissection in fresh cadavers prior to operation permitted resection of 6 cm of trachea, using this technique. They also proposed, where carinal resection was not required, to close the left main bronchial stump. Eschapasse and colleagues used this technique in 1961.119 Archer and colleagues similarly excised a granular cell myoblastoma at the carina in 1963.120 The
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procedure was a major step in carinal surgery. Grillo and colleagues described resection of the carina and trachea for a length of 4 cm to remove adenoid cystic carcinoma, in 1963.121 The right hilum was mobilized and the trachea anastomosed to the right main bronchus. The left main bronchus reached the trachea easily enough to be anastomosed there end-to-side. The patient did well. Cross-field intubation and ventilation were used. Temporary occlusion of the pulmonary artery to the nonventilated lung eliminated shunting, but later rarely proved to be necessary. In 1966, Mathey and colleagues reported results in 7 patients, who underwent carinal excision with or without bronchial resection, using thoracotomy.40 They believed, however, that sternotomy might be preferable. Three patients had pneumonectomy, and 2 had partial lung resection. The following anastomoses were done: trachea to main bronchus; side-to-side left main and intermediate bronchus and both end-to-end to trachea; 2 patients had dermal graft patches. There were 2 postoperative deaths. Eschapasse and colleagues, in 1967, cited 3 patients who had circumferential resection of the entire carina with primary reconstruction.122 Anastomoses were of right main bronchus to trachea with left main end-to-side to bronchus intermedius in 2; left main to trachea with intermediate bronchus to left main. One patient died postoperatively. Eschapasse favored right thoracotomy, cross-table ventilation, avoidance of prostheses, and primary reconstruction (Figure 3). Anesthesia for carinal resection, which had initially seemed formidable, was managed easily enough in patients by cross-table ventilation of the trachea and bronchi as the procedure progressed, so that ventilation was not interrupted or uncontrolled at any point. Nonetheless, the anesthetic challenge of carinal resection suggested the use of extracorporeal circulation to some. Nissen removed a “malignant adenoma” in this way.123 Under bypass, Woods and colleagues excised recurrent “cylindroma” from the carina with very limited margin.124 Reconstruction was by suture and a patch of skin supported by wire mesh. Adkins and Izawa performed lateral resection of the carinal wall for cylindroma, patching the defect with Marlex and mediastinal tissues.125 As might be expected, granulation tissue formed at the patch site. The considerable potential for hemorrhage due to the need for heparinization during bypass was not encountered in these technically limited cases. Experience with carinal resection and reconstruction grew slowly. In 1974, Eschapasse collected 19 cases from several French teams, Perelman and Koroleva recorded 29 carinal resections with reconstruction in 1980, and Grillo had performed 36 carinal reconstructions by 1982.99,126,127 Twenty-three of Grillo’s group were primary tracheal neoplasms, 5 were bronchogenic carcinomas, and 8 were inflammatory lesions. Eleven were reconstructed without loss of lung tissue. On the basis of this experience, Grillo presented a comprehensive schema for carinal reconstruction.99 For short resections, carinal restoration was by side-to-side main bronchial anastomosis, which was then joined end-to-end to the trachea; for longer lesions, the trachea was placed end-to-end to the left main bronchus (if the gap was less than 4 cm) and the right main bronchus endto-side to the trachea; for still more extensive tracheocarinal removal, the “Barclay” anastomosis of the right main bronchus to the trachea and the left main bronchus end-to-side to the intermediate bronchus was used. Other special problems were also presented, including the problem of lesions involving a long length of trachea and also of the left main bronchus. Recent exploration of problems with carinal reconstruction has updated this experience in 143 resections.128 Approach for carinal resection via right thoracotomy has been preferred by most surgeons.99,121,126,129,130 Left thoracotomy with subaortic dissection was employed for specific lesions, principally those involving the left main bronchus and the carina, but little of the tracheal length.40,99,126,131,132 Left thoracotomy with retroaortic dissection was also explored early, but failed to gain acceptance.116,118 Median sternotomy for carinal access was described in 1907 by Goeltz for foreign body removal, in 1960 by Padhi and Lynn for bronchopleural fistula, in 1961 by Abruzzini for treatment of postpneumonectomy tuberculous fistulae, and was reintroduced with anterior and posterior pericardial opening by Perelman (Figure 4).130,133–135 Pearson and colleagues favored this approach for cari-
Development of Tracheal Surgery: A Historical Review
3 Henry Eschapasse, MD, Chief of Thoracic and Cardiovascular Service Emeritus, Regional Hospital Center of Toulouse, and Professor Emeritus, University of Toulouse. In the decades post World War II, there was great interest and activity in tracheal surgery and pathology in France. Dr. Eschapasse was a leader in this field, and especially interested in the study of primary tracheal neoplasms and carinal reconstruction. Toulouse became a center for tracheal surgery.
FIGURE
nal resection.136 Maeda and colleagues added left anterior thoracotomy to sternotomy to improve access.132 Grillo employed bilateral thoracotomy (“clamshell” incision) for free access to the carina and to both thoraces for treatment of complex lesions, especially those involving the left main bronchus, carina, and a long extent of the lower trachea.99
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FIGURE 4 Mikhail I. Perelman, MD, Consulting Surgeon, National Research Center of Surgery, Moscow, and Professor of Surgery and Physiopneumonology, Moscow Medical Institute. Professor Perelman had an early interest in airway surgery, acquired a large clinical experience, and published the first comprehensive books in the field. Tracheal tumors were a special interest of his.
Carinal Pneumonectomy Surgeons early conceived of extension of pneumonectomy for bronchogenic carcinoma to include tumors that also involved the carina.111,113,137 Carinal pneumonectomy with anastomosis of the terminal end of the trachea to the left main bronchus was reported in 1958 by Mattes and in 1959 by Gibbon.76,138 Hardin and Fitzpatrick, in 1959, excised the carina for bronchogenic carcinoma, reconstructing the carina by direct suture with the aid of free cartilage graft, using ventilatory anesthesia delivered by a tube placed in a distal left main bronchial aperture.139 The graft was taken from an uninvolved portion of the right bronchus.
Development of Tracheal Surgery: A Historical Review
MacHale did the same in 1972.140 In 1966, Thompson described anastomosis of the left main bronchus to a tailored trachea after right pneumonectomy for squamous cell carcinoma, which included a sleeve of carina.141 Also in 1966, Mathey and colleagues expressed a preference for circumferential carinal resection over lateral resection and patching.40 Grillo included carinal pneumonectomy with circumferential resection in the spectrum of techniques of carinal resection and reconstruction.99 Carinal pneumonectomy for bronchogenic carcinoma became further established with significant series reported by Jensik in 1972, Ishihara in 1977, Deslauriers in 1979, Dartevelle in 1988, Tsuchiya in 1990 and their colleagues, and Mathisen and Grillo in 1991.142–147 The initially high operative mortality of nearly 30% proved to be largely due to a form of acute respiratory distress syndrome labelled postpneumonectomy pulmonary edema, of noncardiogenic origin. Mathisen and colleagues showed favorable response to prompt treatment with nitric oxide.148 Believed to be the result of barotrauma, this dread complication has reduced in incidence with close attention to ventilatory volumes and pressures, so that mortality has fallen to about 10% or lower.145,149,150
Anesthesia for Tracheal Surgery McClish and colleagues noted that concern about anesthesia for major airway reconstruction “stems from the complexity of simultaneously controlling the airways, maintaining satisfactory gas exchange, and ensuring good surgical exposure to the trachea.”151 The technique of ventilation across the operative field, with direct insertion of endotracheal tubes into the trachea and bronchi during phases of surgery when the airway is open, developed early and is described with variations in reports of tracheal reconstructions cited earlier. Tubes across the operative field were used in experimental work and Ravitch early mentioned clinical usage.42,103,104,152 In 1951, Friedmann and Emma described a catheter insufflation technique for carinal resection in one patient.153 Grigor and Shaw, working with Barclay and his colleagues, used crossfield ventilation in combination with endotracheal intubation, depending on intermittent ventilation during the implantation of the left main bronchus into the bronchus intermedius.154 They recognized that the preceding development of one-lung anesthesia provided the groundwork for carinal anesthesia. Baumann and Forster, in 1960, described systematic approaches to anesthesia for cervical, intrathoracic, and carinal tracheal surgery, including intubation via distal tracheostomy and also across the operative field.155 Grillo and colleagues, in 1963, detailed similar technique for carinal resection and reconstruction.121 Cross-table anesthetic techniques were fully described by Grillo in 1965 and expanded in 1970.83,129 The potential use of two anesthesia machines for complex carinal reconstruction was also noted. Geffin and colleagues summarized cross-table anesthetic techniques for tracheal and carinal reconstruction in 1969 on the basis of their accumulated experience by then with 31 operations.156 Theman and colleagues confirmed these techniques.157 Lee and English, in 1974, described the use of a Saunders-type bronchoscopic injector through a catheter passed beyond a tracheal stenosis.158 El Baz and colleagues favored high-frequency positive pressure ventilation for tracheoplasty to permit better visualization and access.159 McClish and colleagues expressed similar convictions in a series of 18 patients.151 Wilson thoroughly updated these anesthesiologic approaches.160 Although his preference was for cross-table ventilation, high-frequency ventilation was valued as a useful adjunctive technique in special circ*mstances, such as complex carinal reconstruction. These choices remained a matter of preference, with both techniques proving satisfactory in experienced hands. All agree that close communication and cooperation between surgeon and anesthesiologist are uniquely demanded for this type of surgery, preoperatively, intraoperatively, and, optimally, postoperatively. The use of cardiopulmonary bypass for tracheal and especially carinal resection has been mentioned earlier. In their extensive experiences with tracheal surgery, Eschapasse, Grillo, Pearson, and Perelman found bypass to be unnecessary. Its use in very complicated cases, where it might be theoretically desirable,
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but where extensive dissection and manipulation of the lung was required, led to fatal pulmonary parenchymal hemorrhage in 2 patients due to necessary anticoagulation (Grillo, Pearson, unpublished).
Laryngotracheal Resection and Reconstruction Just as reconstruction of the carina presented unusual difficulties, so did the proximal end of the airway. When tracheal lesions also affect the subglottic larynx, the anatomic and functional characteristics of that structure offer special problems. Many otolaryngologic procedures were developed to manage inflammatory stenosis at this level, when conservative measures failed. The latter measures included dilation, stents, intubation, steroid injection, cryotherapy, and laser surgery. Surgical procedures that were devised included anterior and posterior cricoid splits, placement of stents, mucosal and cutaneous grafts, free grafts of cartilage and hyoid, pedicled hyoid, cutaneous flaps variously supported with cartilage, and multistage “trough” procedures. These many operations will not be reviewed here, but, in general, success was limited.161 A one-stage approach to subglottic stenosis characterized by cricoid involvement developed slowly. The initial work was done by otolaryngologists, but full development of the techniques was accomplished by thoracic surgeons who faced the problem of subglottic stenosis as it presented in the spectrum of post intubation tracheal stenosis. Conley removed the entire cricoid in 1953 for a chondroma, preserving the mucoperichondrium, which was held in place by a foam rubber stent.67 Great care was taken to avoid injury to the recurrent laryngeal nerves. Shaw and colleagues resected damaged or stenotic cricoids in 2 patients with anastomosis to the thyroid cartilage.27 Existing vocal cord paralysis simplified the problem in these patients. Ogura and Roper apposed the second tracheal ring to thyroid cartilage after subtotal excision of traumatically scarred and stenotic cricoid in 2 patients.94 The recurrent nerves were paralyzed, arytenoidectomy was done, and a stent was used postoperatively. The distal trachea was mobilized and the thyrohyoid muscles and constrictors, which are attached to the thyroid cartilage, were divided to assist in approximation. Subperichondrial cricoid resection avoided injury to the recurrent nerves.95 Six of 7 patients with chronic subglottic stenosis were helped by this procedure. In 1974, Gerwat and Bryce placed the upper line of resection for stenosis at the lower border of the thyroid cartilage anteriorly, and through the posterior cricoid lamina below the cricothyroid joints posteriorly.162 Thyrohyoid release was added and believed to be important. Four patients were so treated. In 1975, Pearson and colleagues followed the same line of cricoid resection, but rongeured all but a thin shell of posterior lower cricoid plate, sutured the ends of the first intact cartilaginous ring of trachea together, and inset this into the rongeured groove to form the laryngotracheal anastomosis (Figure 5).163 Recurrent nerves were preserved. Superior laryngeal release was done, and a splinting T tube was added postoperatively. Six patients were successfully treated. Couraud and colleagues, in 1979, added 4 patients, all but one successful (Figure 6).164 They pointed out that there was no use in disturbing the recurrent nerves, that sometimes the posterior cricoid cartilage did not need to be tailored, and that tracheostomy was not regularly necessary. Grillo, in 1982, described 18 patients with subglottic stenosis treated with a somewhat modified procedure.161 In patients with anterolateral stenosis, a simple bevelled cricoid resection was sufficient, and the tracheal cartilage to be anastomosed was obliquely tailored to fit easily. For circumferential stenosis, scar over the posterior cricoid plate was excised and the raw area resurfaced with a broad-based flap of posterior membranous tracheal wall shaped for this purpose. Neither laryngeal release nor tracheostomy was routinely needed. In 1992, Grillo and colleagues reviewed 80 patients who underwent one-stage laryngotracheal resection and reconstruction for subglottic stenosis by these techniques: 50 with postintubation lesions, 7 from trauma, 19 idiopathic, and 4 others.165 Thirty-one patients required circumferential resection with posterior flap resurfacing. There were 2 failures. If glottic correction was also needed, it was done initially as a separate procedure. Maddaus, with Pearson’s group, proposed synchronous glottic reconstruction where that was
Development of Tracheal Surgery: A Historical Review
5 F. Griffith Pearson, MD, Chief of Thoracic Surgery and Surgeonin-Chief Emeritus, Toronto General Hospital, and Professor of Surgery Emeritus, University of Toronto. Dr. Pearson, who founded and led the Thoracic Surgical Division in the Toronto General Hospital, early became interested in tracheal surgery. He contributed richly to the understanding and treatment of postintubation stenosis, to the development of one-stage laryngotracheal reconstruction, and to our knowledge of adenoid cystic carcinoma.
FIGURE
also required, reporting 15 such cases of 53 subglottic repairs.166 They also adopted the posterior tracheal membranous wall flap described by Grillo and his colleagues.161,165 Monnier and colleagues proved this type of repair to be useful in children.167
Cervicomediastinal Exenteration and Mediastinal Tracheostomy Rarely, following extensive resection of the larynx and upper trachea for neoplasms, such as thyroid carcinoma, adenoid cystic carcinoma, or recurrent laryngeal carcinoma after laryngectomy, there is need for mediastinal tracheostomy, well below the sternal notch. Watson, in 1942, devised a procedure to treat a patient with squamous carcinoma 4 cm above the carina.168 The patient had undergone laryngectomy for cancer 15 years earlier, followed by radium treatment. A “V” of sternum was resected and skin flaps mobi-
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6 Louis Couraud, MD, Chief of Thoracic Surgery, Emeritus, Xavier Arnozan Hospital, Pessac, and Professor of Surgery Emeritus, University of Bordeaux II. Professor Couraud made Bordeaux renowned for airway surgery, producing excellent surgical results and adding to our knowledge of many aspects of tracheal disease: postintubation stenosis, laryngotracheal stenosis, juvenile tracheal growth, tracheoesophageal fistula, primary tumors, postsurgical complications, and the airway in transplantation.
FIGURE
lized to allow closure of the margins of the tracheal stoma. In 1951, Sloan and Cowley managed the problem of tracheal compression by an aortic aneurysm by establishing a side tracheostomy, the tube of which emerged from the back medial to the right upper scapula, after removal of proximal rib segments.169 After wrapping the aneurysm, it was possible to remove the tube. The authors discussed earlier proposals and even attempts to establish transpleural bronchial fistulae for this purpose, and a proposal, not acted upon, by Gluck in 1907, to perform posterior bronchotomy. In 1952, for mediastinal tracheostomy, Kleitsch removed the upper sternum and inserted a polythene tube.170 A sequence of irradiation necrosis and recurrent tumor frustrated plans to line the opening with skin grafts. In the same year, Minor, after removal of recurrent carcinoma of the tracheal stoma, brought skin flaps as a tube through a sternal opening to connect with the trachea.171 Healing failed, and the patient bled to death 4 months later. Waddell and Cannon, in 1959, pulled a short tracheal stump to the right of
Development of Tracheal Surgery: A Historical Review
the ascending aorta and created a skin tube from crossed anterior chest skin flaps which passed through a hole rongeured in the sternum and was anastomosed to the tracheal end.172 Two of 4 patients, all with squamous cell carcinoma, died of massive hemorrhage. In 1962, Sisson and colleagues, operating for recurrent laryngeal carcinoma at the tracheal stoma, excised a large portion of surrounding skin with the specimen and removed the manubrium and the heads of the clavicles.173 Skin flaps were turned up to effect closure about the stoma, and an inferior defect was skin grafted. After 2 patients died from innominate artery hemorrhage postoperatively, the pectoralis muscles were undermined and rotated between the innominate and left carotid arteries and the trachea. Also in 1962, Ellis and colleagues used a tube of heavy Marlex mesh to reach the surface after low transection of the trachea.174 Granulation tissue formation and the possibility of infection, erosion, and hemorrhage make tubes of foreign material undesirable in this setting. In an effort to eliminate tension at the tracheal cutaneous anastomosis, which carried the threat of subsequent nonhealing and fatal innominate hemorrhage, Grillo in 1966 proposed fashioning a broad full-thickness bipedicled flap of anterior chest wall skin and subcutaneous tissue formed with two long, horizontal incisions (Figure 7).175 This flap was depressed to meet the end of the trachea in the mediastinum, made accessible by resection of manubrium, heads of clavicles, and upper two costal cartilages. The stoma emerged in midflap, resulting in a simple suture line more likely to heal well. Two end stomas and one in-continuity stoma were reported. Grillo and Mathisen subsequently offered further guard against vessel erosion in the event of deficits in peristomal healing by 1) advancing omentum routinely to separate trachea and great vessels, and 2) electively dividing the brachiocephalic artery under electroencephalographic monitoring, where the tracheal stump was very short, following preoperative angiography.176 One operative death occurred in 18 patients. Additional experiences have been recorded in this area by Stell, Krespi, Gomes, and Orringer and their colleagues.177–180 Withers and colleagues suggested use of a pectoralis musculocutaneous flap, which has particular application to cases where a wide resection is necessary around an existing stoma for reason of peristomal carcinoma or irradiation damage.181
Complications of Tracheal Surgery As tracheal surgery became more common, a pattern of complications inevitably appeared. These were analyzed by Levasseur in 1971, Couraud in 1982, and by Grillo in 1986, along with their colleagues.92,182,183 In 11 patients, Levasseur and colleagues observed 1 restenosis and 4 lethal erosions of brachiocephalic artery.92 This led the authors to recommend cervical muscle or thymic interposition for prophylaxis against hemorrhage. Couraud and colleagues, reporting on 122 cases of resection with only 4 deaths and 1 failure, emphasized anti-infectious and anti-inflammatory precautions. 182 Grillo and colleagues reviewed incidence, causes, treatment, and prevention of complications in 416 primary reconstructions, 279 for postintubation lesions.183 Suture line granulations occurred in 28 of 209 cases, when various nonabsorbable sutures were used for anastomosis, but in none of 113 after the adoption of absorbable Vicryl sutures. Brachiocephalic artery injuries were best avoided by avoiding any dissection of the artery, and where that was not possible, interposing a strap muscle. From the first to the second halves of the series, deaths fell from 4 to 1, failures from 13 to 7, and complications from 42 to 30.
The Unreconstructible Trachea: T Tubes and Stents T tubes had been devised and variously employed in the past, as by Bond in 1891 and Falbe-Hansen in 1955, citing Laurens’ earlier use of a T tube.184,185 Aboulker and colleagues treated postintubation injuries with a T tube in 1960.186 The silicone rubber T tube developed by Montgomery in 1965 proved widely useful in tracheal surgery, although it was developed initially in the false hope that prolonged stenting would resolve
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FIGURE 7
Hermes C. Grillo, MD, Chief of General Thoracic Surgery, Emeritus and Senior Surgeon, Massachusetts General Hospital (MGH), and Professor of Surgery Emeritus, Harvard Medical School. The picture shows Dr. Grillo commencing a cervicomediastinal exenteration in 1966. His first assistant is Mortimer J. Buckley, then Chief Resident in Surgery at MGH, later to become Chief of Cardiac Surgery.
tracheal stenosis.187 Cooper and colleagues in 1981 and Gaissert and colleagues in 1994 used it for permanent and temporary restorations of airway continuity when the trachea was not reconstructible, a lesion was not removable, or a temporary airway was needed.188,189 Westaby and colleagues added a bifurcated T tube for help in carinal problems.190 The development and deployment of stents will not be reviewed here. However, caution needs to be raised against tendencies to use essentially permanent expandable stents where lesions might otherwise be readily and definitively corrected by surgery. The result too often is doubly negative: correction of the lesion is permanently prevented and severe complications may develop from the stent.191 Removable silicone
Development of Tracheal Surgery: A Historical Review
stents also hinder curative treatment and may quite readily cause granulations, especially in the subglottic region. These are sometimes reversible, however, in contrast to problems caused by permanent stents.
Treatment of Tracheal Diseases Primary Tracheal Tumors Thus far, this review has focused on the evolution of techniques of tracheal surgery. Application of these and other additionally developed techniques to specific diseases of the airways will now be considered. The challenge of treating the rare tracheal tumors which were seen provided the initial stimulus for tracheal resection.1,5 The very rarity of primary tracheal neoplasms, on the other hand, provided limited incentive to attack this problem systematically. In 1938, Culp collected 433 reported cases of primary tracheal tumors, beginning with Lieutaud’s discovery of fibroma at autopsy in 1767.63 From prior cumulative series, Culp noted the slow increment from 147 cases in 1898, to 201 in 1908, to 252 in 1914, and 351 in 1929. He provided an exhaustive bibliography, but personally found only 1 carcinoma in 9,000 autopsies at McGill University, and 1 in 12,700 autopsies at Montreal General Hospital. Ellman and Whittaker raised the total to 507 in 1947.64 “Cylindroma” was often classified as adenocarcinoma, and tracheopathia osteoplastica was included as a tumor. Houston and colleagues collected 53 primary cancers of the trachea in over 30 years at Mayo Clinic, showing a distribution now recognized as expected: 45% squamous, 36% “cylindroma” (adenoid cystic carcinoma), and the balance of other origins, including mesenchymal tumors.192 Reporting a 30-year experience in 1969, only 2 squamous cancers had been removed, 1 by lateral excision, 6 adenoid cystic (none by circumferential resection and anastomosis), and 1 mucoepidermoid by end-to-end repair. The next year, Hajdu and colleagues described 41 patients with primary tracheal carcinoma from Memorial Hospital over 33 years: 30 squamous and 7 adenoid cystic.193 Few were treated by resection. Times were changing, however, as techniques of resection based on anatomic mobilization were increasingly applied to tracheal neoplasms. Forster and colleagues resected a cervical tracheal epithelioma in 1957 with end-to-end suture.71 Forster and Holderbach published a voluminous report in 1960 on the pathology and clinical presentation of tracheal tumors, and of experimental and a few clinical trials at that early date.194 Non-neoplastic lesions were also included. Grillo recounted treatment of three primary tumors by circumferential resection in 1965, using cross-table ventilation through the open trachea.83 Mathey and colleagues reported resecting 5 patients with primary tracheal neoplasms in 1966, with 1 early and 1 late postoperative death.40 Perelman and Korolyova successfully treated 5 patients with primary tracheal intrathoracic cancer by circular resection and anastomosis in 1968.87 They introduced an anesthesia tube into the left main bronchus via an incision in the membranous wall of the right main bronchus. Dor and colleagues reported in 1971 on resections of tracheal tumors in 6 patients, with 3 postoperative deaths.91 By 1973, Grillo had excised 11 primary tumors and 5 secondary tumors in a series of 100 tracheal resections with reconstruction.195 Nine of the 11 patients were alive without disease; 1 patient died postoperatively. Experience with surgical management began to grow. In 1974, Eschapasse reported on 152 patients with primary tracheal tumors treated by 12 French and 2 Russian groups, which included 32 circumferential resections and 18 carinal reconstructions.126 The poorest long-term results were with squamous carcinoma, which also gave the highest postoperative mortality. Adenoid cystic carcinoma showed prolonged survival, but late recurrence. Also in 1974, Pearson and colleagues accomplished 5 resections of adenoid cystic carcinoma with primary anastomosis, without postoperative death.196 In 6 others, prosthetic replacement was made using Marlex. Grillo reported seeing 63 patients with tumors by 1978.85 Nineteen patients with primary tumors (and 5 more with secondary) were treated by cylindrical resection and anastomosis and 10 additional patients by carinal resection and reconstruction. Ten others underwent staged reconstruction, and 10 had laryngotracheal resection, or were treated by other means. Two died after cylindrical resection and 3 after carinal resection and reconstruction.
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Subsequent major series began to define the long-term oncologic expectation. In 1990, Grillo and Mathisen reported the largest single institutional series of 198 primary tumors treated at Massachusetts General Hospital (MGH) over 26 years.6 Resection rates were 63% for squamous carcinoma and 75% for adenoid cystic carcinoma. Pearson and colleagues updated their experience with 44 tracheal tumors in 1984 and a subsequent report by Maziak and colleagues did the same for 38 adenoid cystic carcinomas in 1996.136,197 Regnard and colleagues collected 208 patients in a multicenter series in France in 1996.198 In that same year, Perelman and colleagues summarized an experience with 144 primary tumors.199 Squamous carcinoma and adenoid cystic carcinoma together compose about 75% of all primary tracheal tumors, in comparable numbers. The etiology, curability, and associated aerodigestive carcinomas of squamous cancer were much like squamous lung cancer. Surgery for adenoid cystic carcinoma, combined with radiotherapy, produced high 5-year survival rates, but a continued fall in survival at 10 years and thereafter occurred, due to local recurrence and the appearance of metastases. The rarity and idiosyncratic and prolonged course of adenoid cystic carcinoma clearly requires very prolonged observation for complete clinical definition. An enormously wide variety of tumors of other histology, most often benign or of low-grade malignancy, composed the remaining 25% of cases.6 Operative mortality in all patients ranged from 5.3 to 10.5% in various series. Mortality and morbidity fell with surgical experience, but remained highest in carinal reconstruction.183
Secondary Tracheal Tumors Resection of the carina for bronchogenic carcinoma has been discussed under the section, “Carinal Pneumonectomy,” above. The proximity of the thyroid gland makes the trachea and lower larynx targets susceptible to invasion by cancer in this gland.200 Localized tracheal invasion by thyroid neoplasms was resected episodically as tracheal surgery evolved. Rob and Bateman, in 1949, resected six rings of trachea and a portion of cricoid for recurrence of thyroid cancer “of low malignancy,” 7 years after initial excision and radiotherapy.30 Tantalum gauze-fascia lata reconstruction was done, leaving a strip of posterior mucosa. After a checkered course, the patient survived. Conley did a staged repair with tantalum mesh and fascia plus skin flaps after resection of anterior tracheal wall invaded by “adenocarcinoma of the thyroid.”67 Lazo resected the anterior wall of the cervical trachea for thyroid cancer in 1957, using a prosthesis for speech.201 In 1965, Grillo resected a six-ring segment of trachea, including a portion of cricoid invaded by papillary carcinoma that had paralyzed the left cord and obstructed the tracheal lumen.83 Tracheal reconstruction was staged with a cutaneous tube supported by inlying polypropylene rings. The result was satisfactory. In 1966, Mathey and colleagues resected 3.5 cm of trachea for papillary carcinoma and performed an end-toend anastomosis, but placed a tracheostomy in the suture line postoperatively.40 An aggressive approach was accepted early in Japan, but only slowly in the west. Ishihara and colleagues reported operation on 11 patients in 1978, 8 of whom had recurrent papillary adenocarcinoma after prior surgery.202 Sleeve resections were done with resection of the anterior cricoid in 3 of the patients. Two died from operation and 3 developed laryngeal stenosis. Five were long-term survivors. This same group reported on 60 patients by 1991.203 In 1985, Tsumori and colleagues reported 18 resections with anastomosis.204 In 1986, Fujimoto and colleagues performed sleeve resection in 6 patients and window resection in 3.205 A survey of tracheobronchial surgery in Japan, reported in 1989 by Maeda and colleagues, revealed 151 cases of tracheoplasty for thyroid cancer against 147 tracheobronchial tumors over a period of 30 years.206 In the west, Grillo listed 3 patients in 1978, resected for thyroid carcinoma, and recommended that this treatment be applied more widely.85 In 1986, Grillo and Zannini cited 16 patients treated by resection and reconstruction, and by 1992, Grillo and colleagues reported 27 cases.207,208 Rationale for this approach is adherence to the oncologic principle of thyroid surgery, that local disease be removed totally. The surgery is not highrisk surgery or radical surgery in competent hands. Given the proclivity of papillary tumors to become more
Development of Tracheal Surgery: A Historical Review
aggressive in time, plus the observation that many of these patients had undergone “shave” procedures, often years before, anything less than complete removal (including airway, if necessary) seems inappropriate. Nonetheless, “shave” procedures in the case of superficial invasion, and “window” resection in the case of deep invasion, are still being recommended by surgeons without extensive experience in tracheal reconstruction.209 Radical extirpation of invasive undifferentiated thyroid carcinoma and also of massive recurrences of papillary carcinoma, to include laryngectomy and extended tracheal resection, was described by Hendrick, in 1963, in 11 patients with 5 long-term survivors and 5 alive without disease from 4 to 16 years.210 In 1958, Frazell and Foote noted 3 of 4 patients, who had laryngeal and tracheal resection for thyroid cancer, lived 41⁄2 to 5 years.211 Grillo and colleagues reported in 1992 on radical extirpation of tumor in 7 patients, by cervicomediastinal exenteration including esophagectomy.208 Palliation is a principal goal of these procedures.
Postintubation Lesions The poliomyelitis epidemics of the mid-twentieth century introduced and led to an ever-widening use of mechanical ventilators to treat respiratory failure. The iatrogenic lesions that resulted provided a whole new field of endeavor for the tracheal surgeon. Gradually, a spectrum of lesions was recognized, attributable to ventilatory apparatus: endotracheal and tracheostomy tubes and the cuffs necessary to seal the trachea.84,90,186,212 Principal among these were 1) circumferential stenosis that appeared at the level of the sealing cuff, and 2) anteriorly pointed, arrow-shaped stenosis, which occurred at the stomal level. Additionally, granulomas occurred at the point where a tube tip impinged on the tracheal wall. Areas of malacia were seen less often at the level of the cuff and sometimes in the segment between a tracheal stoma and a cuff stenosis. Tracheoesophageal fistulae occurred principally between the trachea and the esophagus at the cuff level, usually with accompanying circumferential tracheal damage. Rare, but disastrous when they occurred, were tracheal innominate artery fistulae. These lesions proved to be of two types: one where a tracheostomy tube rested immediately on the innominate artery near the stoma, and another, where the cuff, or, even less often, the tube tip, eroded through the trachea anteriorly into the innominate artery. In the 1960s, numerous papers, often single case reports, appeared in Europe and North America, describing surgical resection of postintubation strictures. Included among these together with their colleagues were Forster in 1957, Flavell in 1959, Witz in 1960, Binet and Aboulker in 1961, Van Wien in 1961, Mathey in 1966, Byrn as well as Fraser in 1967, and Jewsbury, Dor, Dolton, Schaudig, Lindholm, and Naef in 1969.40,71,73,75,88,213–221 Series of cases also were reported by the following authors together with their colleagues: Deverall reported 6 patients in 1967, Pearson reported 15 in 1968, Grillo reported 14 whereas Couraud reported 9 in 1969, and Dor reported 9, Levasseur reported 10, and Harley reported 11 in 1971.84,86,89,91–93,212 These last authors, especially Pearson, Grillo, and Harley, with somewhat broader experience, defined the anatomic and pathologic differences between stomal and cuff stenoses and other postintubation injuries, and discussed their pathogenesis. Malacia instead of stenosis was also described, although a rare finding, by Grillo.222 Deverall, Pearson, Grillo, and Couraud and their colleagues stressed the importance of allowing florid inflammation to subside prior to surgical correction.84,86,89,212 Their generally good results showed the superiority of definitive surgical resection and anastomosis over prior alternative methods of treatment, such as repetitive dilation, steroid injection, or cryotherapy. Unfortunately, the lesson is being relearned today, with uncritical use of laser surgery for these lesions,223 and, more lately, with much more disastrous results, the attempted use of stents to treat postintubation stenosis.191 Postintubation lesions became, and remain, the most common indication for tracheal resection and reconstruction. Generally very good results have been obtained in major cumulative series of patients with iatrogenic tracheal and subglottic laryngotracheal stenosis: Bisson and colleagues achieved an 87.5% “cure” in 200 patients in 1992, Couraud and colleagues reported 96% success in 217 patients in 1994, and Grillo and colleagues cited 94% success in 503 patients in 1995.98,224,225
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Correction of postintubation stenosis involving the subglottic larynx remains more difficult than lesions confined to the trachea. The evolution of procedures for laryngotracheal resection and reconstruction by partial cricoid resection has been outlined and their application to iatrogenic stenosis noted. Monnier and colleagues applied this approach in infants and children, also with encouraging success.167 Reoperative tracheal resection and reconstruction for unsuccessful repair of postintubation stenosis proved to be surprisingly manageable. In 1997, Donahue and colleagues tallied 92% good or satisfactory results in 75 patients who had failed prior surgical repairs, 59 of whom were referred.226 Tracheoesophageal fistulae (TEF) due to erosion by tracheal cuffs and often of “giant” size were noted early, in 1966 by Le Brigand and Roy and several other French surgeons in the same period, by Flege in 1967, and by Hedden and colleagues in 1969.227–229 Scattered attempts of repair by sometimes multistaged techniques, including Braithwaite’s successful use of a cutaneous flap to seal the tracheal side of a large fistula in 1961, did not often meet with success.227,230,231 Grillo and colleagues, in 1976, described a definitive one-stage technique for esophageal closure, tracheal resection (where a circumferential cuff lesion was present), and strap muscle interposition, with good results in 7 patients.232 Postintubation injury, however infrequent, has become the most frequent cause of acquired TEF. It is now effectively managed by the type of procedure noted, and has been further described by Mathisen, Couraud, and Macchiarini and their colleagues.233–235 When the fistula is small and the tracheal lesion is not circumferential, tracheal closure is performed. The techniques developed have been applied effectively to closure of TEF from a variety of causes, including trauma and inflammation. Tracheal innominate artery fistula (TIF), described by Lunding in 1964, Silen and Spieker and Stiles in 1965, Couraud and colleagues in 1966, and Foley and colleagues in 1968, as a consequence of tracheostomy and ventilation, was effectively approached surgically by Grillo in 1976, Cooper in 1977, and Couraud and colleagues in 1984.236–243 The mechanism of fistulization was either erosion of the tracheal wall by a highpressure cuff, angulation of a tracheostomy tube tip, or most commonly, erosion by the tube in a low-lying tracheostomy where the elbow of the tube essentially rests on the artery. Jones and colleagues reviewed the topic extensively in 1976, including delineation of types of erosion, emergency management, safety and desirability of arterial resection, and success rates.244 The etiology of postintubation stenosis and other injuries was initially unclear. Among the factors thought to be implicated were irritation from materials of which tube and cuff were made, elution of chemicals by gas sterilization, age, debility, steroids, bacterial infection, and direct irritation by the tube’s presence. Although some of these likely contributed to the injuries seen, pressure and necrosis from tubes and cuffs, whether endotracheal or by tracheostomy, with subsequent efforts at tissue repair, and, finally, cicatrization, proved to be the fundamental explanation. Post-tracheostomy stenosis had been pointed out as early as 1886, when Colles found four strictures in 57 patients treated for diphtheria.245 However, only with the growing use of ventilation, during and after the 1952 poliomyelitis epidemic, did postintubation injuries become more frequent. In 1960, Aboulker and colleagues identified inflammation as a major factor in the spectrum of posttracheostomy stenosis.186 On the basis of 12 autopsy studies in patients who were ventilated via tracheostomy for differing time periods, Bignon and Chrétien in 1962 described inflammation, metaplasia, and stenosis at the tracheostomy site; pseudopolyps, ulceration, and stenosis in the trachea at cuff level; and, sometimes, softening of the tracheal wall.246 They attributed these changes principally to trauma from the cannula above and to ischemic compression by the cuff or erosion by the tip of the tube below. The severity of lesions did not correlate with the length of ventilation. Yanagisawa and Kirchner as well as Atkins, in 1964, described severe damage to the trachea and stenosis from use of cuffed tracheostomy tubes.247,248 In 1965, after careful autopsy studies of tracheostomized and ventilated subjects, Florange and colleagues reconstructed the evolution of tracheal necrosis from
Development of Tracheal Surgery: A Historical Review
mucosal inflammation to erosion of the mucosa, loss of cartilage, and localized mediastinitis.249 They concluded that this damage could result in stenosis. In 1965, Stiles described severe changes at the stomal, cuff, and tube tip levels in 23 patients in 37 consecutive tracheostomies, all of whom died after ventilation.238 He was inclined to relate the damage to the materials from which the tubes were manufactured. Gibson concluded in 1967 that the “main factors” in producing stenosis were cuff trauma plus infection at the stoma.250 Most tracheae of patients who died while being ventilated via tracheostomy showed necrosis. Murphy and colleagues, in 1966, could only produce stenosis in dogs with cuff tracheostomy when infection was also present.251 In 1968, Foley and colleagues described the tracheal changes due to abutment of tubes and cuffs in patients with fatal burns.240 In 1969, Grillo showed similar changes as a result of ventilation.84 Cooper and Grillo presented a detailed pathologic study of autopsy specimens from patients dying on respirators.252 A spectrum of changes was described similar to that noted by Florange. Lesions appeared within 48 hours and progressed from tracheitis to ulceration of the mucosa, to fragmentation of cartilage, to replacement of the tracheal wall with scar tissue. The location and nature of the lesions also correlated with surgically removed stenotic lesions. Lindholm presented a detailed study in 1969 of lesions developed in the larynx and also in the trachea from ventilation.221 The severity of histologic changes was vastly greater than those described after tracheostomy alone.253 Andrews and Pearson prospectively examined the trachea of 103 patients receiving ventilator support in 1971.254 Twelve stomal and 6 cuff stenoses developed. Bronchoscopic examination was of little value in predicting which patients would go on to stomal stenosis, but circumferential mucosal ulceration at the cuff level dependably predicted stenosis at that level. Additional statistically significant factors observed in this study were large tracheostomy tubes and high-dose steroids. The same erosive processes were observed to cause tracheoesophageal fistulae and tracheoinnominate artery fistulae. Prevention of postintubation injury quickly became a priority once the origin of these lesions was evident. In 1957, Adriani and Phillips found that most of the intracuff pressure necessary to inflate the then conventional cuffs (90 to 220 mm Hg) was expended on distending the cuff, and the pressure on the tracheal wall was low (10 to 15 mm Hg) in order to develop ventilatory pressures of 10 to 20 mm Hg.255 Cooper and Grillo later pointed out that excessive pressures were necessary to seal the irregularly-shaped trachea by distending the relatively rigid small volume cuffs that were then in use.256 Knowlson and Bassett also noted that small increments over the minimal occlusive volume necessary for the seal of conventional cuffs at 20 cm H2O caused a rapid rise in the pressure exerted on the tracheal mucosa.257 In 1943, Grimm and Knight had proposed that the ideal cuff “should have sufficient volume when inflated, without stretching, to fill the diameter of the trachea.”258 Lomholt offered a cuff of thin and elastic Teflon in 1967, lying in folds so that intracuff pressure would be identical with pressure on the mucosa.259 Carroll and colleagues, in 1969, recommended a cuff with large residual volume, a large sealing area, a centered tube, and the development of only small increases in tracheal wall pressure with overinflation.260 Cooper and Grillo reproduced severe stenosing cuff lesions in dogs in 1969, which were entirely parallel with lesions seen in man (Figure 8).256 They used standard balloon cuffs and inflation necessary for ventilation at 20 to 25 cm H2O. Intraluminal pressures were 180 to 250 mm Hg. Experimental largevolume, thin-walled latex cuffs produced seals at 20 to 40 mm Hg intraluminal pressure, and no mucosal damage followed. Since this conclusively proved that tracheal lesions were due to cuff pressure, a largevolume, compliant cuff was designed for clinical use by Grillo and colleagues.261 Forty-five patients were randomly selected for ventilation with a then standard Rusch cuff or the experimental large-volume, compliant latex cuff, and the resulting tracheal injuries were evaluated and compared. Any degree of injury severe enough to evolve into stenosis was produced by the standard (high pressure) cuff. The average intracuff pressure in the new cuff was 33 mm Hg compared with 270 mm Hg in the standard cuff. In extensive clinical use, no tracheal lesions resulted from use of this large-volume, compliant cuff.
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8 Joel D. Cooper, MD, in about 1970, when, as surgical resident at Massachusetts General Hospital, he worked with Dr. Grillo on the etiology and prevention of postintubation cuff tracheal stenosis. Dr. Cooper went on to perform successful lung transplantation at Toronto General Hospital and developed lung volume reduction surgery for emphysema when Chief of Thoracic Surgery at Barnes-Jewish Hospital and Professor of Surgery at Washington University in St. Louis. FIGURE
For economic reasons, manufacturers later abandoned latex in favor of plastic cuffs, which lack extensibility. When overinflated just a bit, the present day large-volume cuffs invariably produce steep rises in intracuff pressure, with the potential for tracheal injury severe enough to result in stenosis.257 Careful attention to cuff inflation and pressures, however, have avoided any incidents of cuff stenosis since 1970 at MGH. A variety of other seals, including prestretched cuffs, flanges, and alternating cuffs, were also proposed as solutions, but they lacked the simplicity and effectiveness of properly used large-volume cuffs. After adopting the lightweight swivel trachea connectors used at MGH, Andrews and Pearson observed a drop in the incidence of stomal stenosis from 17.5% to 6.9%.254 The addition, since then, of a suspension of connecting tubing to avoid leverage of the tube against the tracheal stoma, has essentially eliminated stomal stenosis at the MGH.
Development of Tracheal Surgery: A Historical Review
Elimination of TEF has followed proper use of large-volume cuffs for ventilation, along with avoidance of inlying rigid nasogastric tubes. TIF has all but disappeared with attention to accurate placement of tracheostomy tubes at the level of the second and third tracheal rings and not below, and also, by appropriate use of large-volume, low-pressure cuffs.
Management of Tracheal Trauma Early experiences with tracheal and bronchial laceration and rupture have been described. In 1959, Hood and Sloan listed their 18 experiences with tracheal injuries in a series of 91 tracheobronchial cases from the literature, and these were more commonly of linear lacerations.26 Shaw and colleagues, in 1961, added 9 cervical and 4 intrathoracic tracheal ruptures, recommending primary repair of acute injuries and resection of scar with accurate anastomosis for post-traumatic stenosis.27 Baumann reviewed the limited knowledge about tracheal trauma in 1960, recommending tracheal bronchoscopy in all serious thoracic trauma.262 Ogura and Powers approached traumatic stenosis of the subglottic larynx aggressively in 1964.95 Chodosh as well as Ashbaugh and Gordon and others described laryngotracheal avulsion injuries.263,264 Beall and colleagues presented 23 tracheal injuries in 1967 and favored immediate treatment, advising airway maintenance and reanastomosis where possible.265 Ecker and colleagues described 21 tracheal injuries in 1971, with 18 successfully treated.266 Bertelson and Howitz reported cervical tracheal rupture and perforating wounds in 1972, recommending tracheostomy alone for small wounds.267 Symbas and colleagues by 1976 progressed from tracheostomy alone to repairs of penetrating wounds in a series of 20 patients.268 Grover and colleagues reported experience with a variety of tracheobronchial injuries in 1979.269 Angood and colleagues added to experience in extrinsic trauma to larynx and cervical trachea in 1986.270 In 1987, Mathisen and Grillo reported good results with immediate repair of acute tracheal injuries and also of concurrent esophageal transection in 1 patient, and good results in 16 of 17 chronic patients, 14 with vocal cord paralysis and 4 with esophageal injury.271 They emphasized the importance of airway control acutely, assessment of glottic competence where recurrent nerves may be defunctioned, subsidence of inflammation before repair of old injuries, conservation of tracheal tissue, separation of tracheal and esophageal suture lines, and also that a paralyzed larynx can be made functional by adjustment of the glottic aperture. Couraud and colleagues addressed the especially difficult problem of traumatic disruption of the laryngotracheal junction in 1989, describing 19 patients, with restoration of airway and voice in all.272 In general, results of treatment of both acute and late tracheal injuries are very satisfactory, in accord with these established principles. Many additional studies have expanded since then on blunt and penetrating injuries and on iatrogenic lacerations or ruptures due to intubation. Gaissert and colleagues described principles of management of inhalation burns of the trachea in 1993, recommending a conservative approach and very patient use of T tubes.273 Any surgery required is performed very late, after subsidence of the cicatricial response. The irradiated trachea heals poorly when transected and reanastomosed, particularly with rising doses of irradiation and increased intervals between radiation and surgery. Muehrcke and colleagues showed that improved results may be obtained in these difficult problems by wrapping an anastomosis with pedicled omentum.274 Such anastomoses, however, remain at greater risk for serious complications.
Congenital and Pediatric Lesions Concern as to whether growth would occur following resection and anastomosis of the trachea in infants and small children was early allayed by experiment, although occasional success had also been noted clinically. Kiriluk and Merendino had observed growth of main bronchi after anastomosis experimentally.45 Borrie had found stenosis to occur after excision of more than three tracheal segments in lambs.275 Sorensen and colleagues, in 1971, noted somewhat limited growth in anastomotic sites in puppies, after resection of zero to five
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rings.276 Maeda and Grillo, in 1972, noted only mild narrowing of the anastomotic site in puppies without resection, at full growth (Figure 9).277 They found that, after resection, growth also occurred, but the safe anastomotic tension permitting predictable healing was 58% of that acceptable in adult dogs (1,000 g versus 1,750 g).278 Kotake and Grillo observed in puppies that tracheal “stay sutures” reduced anastomotic tension.279 In 1973, Murphy and colleagues noted unpredictably variable growth at anastomosis in piglets, after resection of only two rings.280 Mendez-Picon and colleagues confirmed anastomotic growth in puppies, in 1974.281 In 1978, Burrington found that cartilage grew continuously by proliferation on the convex surface without specific growth centers.282 Vertical incisions, hence, do not interrupt growth.
9 Mazazumi Maeda, MD, pictured in his research laboratory. In 1970 and 1971, he worked as a Research Fellow in Surgery at Massachusetts General Hospital with Dr. Grillo, precisely describing healing of the juvenile trachea after resection. From the University of Osaka, he went to Shikoku, as Professor of Surgery and Chief of Surgery at Kagawa University. He was a leader in introducing tracheal and bronchial surgery in Japan.
FIGURE
Development of Tracheal Surgery: A Historical Review
Cantrell and Guild classified congenital tracheal stenosis in 1964 and reported a case of resection of what later was termed a “bridge bronchus,” with side-to-side anastomosis.283 Tracheal resection and primary anastomosis in children were explored by Carcassonne and colleagues in 1973, Mansfield in 1980, Nakayama and colleagues in 1982, and Grillo and Zannini, and Alstrup and Sorensen, in 1984.284–288 Couraud and colleagues demonstrated long-term growth of anastomotic scars in 1990, particularly after resection of stenosis and anastomosis.289 Monnier and colleagues showed that single-stage laryngotracheal resection and anastomosis was also applicable in small children.167 This procedure appeared likely to largely replace cartilage graft procedures developed earlier.290 However, the length of many congenital tracheal stenoses prohibited resectional treatment. Kimura and colleagues provided a solution in 1982, by inserting a cartilage patch longitudinally the length of the stenosis.291 In 1984, Idriss and colleagues used pericardium for the same purpose.292 Heimansohn and Jaquiss and their colleagues confirmed the use of pericardium and cartilage insets, respectively.293,294 Although successful in most cases, a considerable incidence of repetitive granulations formed on the mesenchymal patch until epithelization eventually occurred, and in some patients, necrosis of the patch required reoperation or tracheostomy.295,296 Tsang and colleagues, working with Goldstraw, solved the problem with slide tracheoplasty, described in 1989.297 Grillo’s report in 1994, describing 4 successful cases so treated, established the procedure.298 A subsequent publication by Grillo and colleagues, reporting a total of 8 successful patients, 1 of whom was 10 days old, confirmed that satisfactory long-term growth occurred after slide tracheoplasty.299 The procedure corrected a long stenosis by providing a firm reconstruction with tracheal tissue, lined with ciliated epithelium and hence with little tendency to form granulomas, which did not require postoperative intubation for support and (absent left pulmonary artery sling or other cardiac anomaly) did not require cardiopulmonary bypass for surgery. Complete laryngotracheoesophageal cleft was successfully repaired in 1984 by Donahoe and Gee.300
Infectious and Inflammatory Lesions The techniques of tracheal and bronchial reconstruction have been applied successfully to infections such as tuberculosis, histoplasmosis, and mucormycosis, and also to a miscellaneous group of lesions including sarcoid and Wegener’s granulomatosis. These are not individually referenced since no new principles were necessary for their treatment. The “new” techniques replaced the wire-supported dermal grafts pioneered by Gebauer for tuberculous airway strictures.52 Idiopathic laryngotracheal stenosis had been identified in scattered case reports in the 1970s.301–303 Grillo and colleagues presented a series of 49 such patients in 1993, 39 of whom were treated by one-stage tracheal or laryngotracheal resection, with 32 good or excellent results.304 Twenty-six patients had been followed from 1 to 15 years, with extension of fibrosis occurring in only 1 patient. The pathology showed dense collagenous fibrosis with little inflammation. No new surgical principles were involved, but definitive delineation of the condition, its pathology, and surgical treatment were provided for the first time. It is, therefore, discouraging to see a recent report of repetitive laser treatment used in 30 patients, who suffered recurrent progressive stenosis, and of failure in 7 patients who did undergo open operation.305 By 2002, 75 patients have been treated surgically at MGH and all but one successfully decannulated.306 Tracheopathia osteoplastica, a very rare condition characterized by submucosal cartilaginous nodules with calcification, most often involving the entire trachea, but also the main bronchi, is sometimes severely obstructive. It was treated by Mark and Grillo and their colleagues, by tracheoplasty over a T tube, which was later removed.307,308 The operation is based on the fact that all pathologic changes are in relation to the cartilaginous wall, allowing outward hinging of the walls to enlarge the lumen.
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Tracheomalacia appears in many forms. Short segments related to postintubation lesions have been resected, whereas longer segments have sometimes been splinted with external polypropylene rings, or internally with stents or T tubes.222 Expiratory collapse associated with chronic obstructive pulmonary disease was treated by Herzog and by Nissen with posterior membranous wall splinting and quilting, pulling the ends of the splayed, softened cartilaginous rings into a more normal C shape.309–311 Thin bone slabs, fascia lata, and later Goretex were used as splint materials. Rainer and colleagues reported results with perforated plastic splints of several designs in 1968.312 Wright and colleagues found posterior splinting of intrathoracic trachea and both main bronchi with strips of Marlex to be effective.313 Importantly, Marlex becomes permanently incorporated by scar tissue, preventing recurrence. The principal clinical benefit is the improved ease in raising secretions.
Postpneumonectomy Syndrome Severely symptomatic airway compression caused by extreme mediastinal shift and rotation after right pneumonectomy was especially noted in children, but has since been identified quite commonly in adults. Its occurrence remains unpredictable. The same effect was observed by Maier and Gould in 1953, in patients with agenesis of the lung.314 The phenomenon was also recognized to occur following left pneumonectomy with a right aortic arch, and, rarely, even after left pneumonectomy with a normal aortic arch.315–318 In 1949, Johnson and colleagues suggested filling a hemithorax with lucite balls to prevent “overdistension” of a remaining lung.319 Adams and colleagues used this technique in a symptomatic child.320 In 1966, Kaunitz and Fisher proposed continued refills of air to maintain normal mediastinal position following pneumonectomy.321 Powell and colleagues used a prosthesis preventively in 1979.322 In 1978, Szarnicki and colleagues divided the aortic arch after placing a graft between the ascending and descending aorta in order to relieve compression.323 Wasserman and colleagues successfully used Silastic breast implants to correct the problem in 1979.324 Rasch and colleagues placed an expandable prosthesis in an infant therapeutically in 1990.325 Grillo and colleagues produced the first report of a series of any size, involving 11 adult patients, in 1992.316 Ten underwent repositioning with implants. Good results were obtained generally, but not in 4 who showed severe residual tracheobronchial malacia after mediastinal repositioning. Malacia was more likely to be present in patients with a very long interval between pneumonectomy and operation. Interestingly and encouragingly, since that report, further severe malacia has not been encountered. Saline-filled breast prostheses are presently successfully employed.
Conclusion Tracheal surgery has largely developed and matured in the last 40 years. In summary, using mobilization procedures, present surgical techniques permit resection of approximately half of the adult trachea with reconstruction by primary anastomosis. Proven methods are also available for laryngotracheal as well as carinal resection and reconstruction. The daunting problem of long congenital tracheal stenosis seems largely solved. Much has also been learned in these decades about the etiology, natural history, pathology, and, in some cases, prevention of various tracheal diseases. The principles of tracheal repair differ little from those of all surgery: accurate diagnosis, thoughtfully designed procedures, refined anesthesia, meticulous and gentle dissection, preservation of blood supply, precise reconstructive technique, scrupulous avoidance of excessive anastomotic tension, protection of suture lines and major vessels by tissue interposition, and avoidance of trauma to fresh anastomoses. In 1960, Baumann and Forster wisely counselled that the simplest solution was likely to be the best, and that the unnecessary sacrifice of even half a centimeter of trachea might force a change in surgical plan.77
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promise after inhalation injury: complex strictures of larynx and trachea and their management. Ann Surg 1993;218:672–8. Muehrcke DD, Grillo HC, Mathisen DJ. Reconstructive airway surgery after irradiation. Ann Thorac Surg 1995;59:14–8. Borrie J. Tracheal stenosis in infancy. Thorax 1960; 15:64–9. Sorensen HR, Moesner J, Nielsen PA. Influence of growth upon the development of stenosis following experimental resection of the trachea in puppies. Scand J Thorac Cardiovasc Surg 1971;5:202–5. Maeda M, Grillo HC. Tracheal growth following anastomosis in puppies. J Thorac Cardiovasc Surg 1972; 64:304–13. Maeda M, Grillo HC. Effect of tension on tracheal growth after resection and anastomosis in puppies. J Thorac Cardiovasc Surg 1973;65:658–68. Kotake Y, Grillo HC. Reduction of tension at the anastomosis following tracheal resection in puppies. J Thorac Cardiovasc Surg 1976;71:600–4. Murphy DA, Dunn GL, Poirier N, Martin M. Growth of tracheal anastomoses: an experimental study in weanling pigs. Ann Thorac Surg 1973;16:158–62. Mendez-Picon G, Hutcher NE, Neifeld V, Salzberg AM. Long term study of tracheal growth after segmental resection in puppies. J Pediatr Surg 1974;9:615–9. Burrington JD. Tracheal growth and healing. J Thorac Cardiovasc Surg 1978;76:453–8. Cantrell JR, Guild HG. Congenital stenosis of the trachea. Am J Surg 1964;108:297–305. Carcassonne M, Dor V, Aubert J, Kreitman P. Tracheal resection with primary anastomosis in children. J Pediatr Surg 1973;8:1–8. Mansfield PB. Tracheal resection in infancy. J Pediatr Surg 1980;15:79–81. Nakayama DK, Harrison MR, deLorimier AA, et al. Reconstructive surgery for obstructing lesions of the intrathoracic trachea in infants and small children. J Pediatr Surg 1982;17:854–68. Grillo HC, Zannini P. Management of obstructive tracheal disease in children. J Pediatr Surg 1984; 19:414–6. Alstrup P, Sorensen HR. Resection of acquired tracheal stenosis in childhood. J Thorac Cardiovasc Surg 1984; 87:547–9. Couraud L, Moreau JM, Velly JF. The growth of circumferential scars of the major airways from infancy to adulthood. Eur J Cardiothorac Surg 1990;4:521–6. Fearon B, Cotton R. Surgical correction of subglottic stenosis of the larynx in infants and children. Ann Otol Rhinol Laryngol 1974;83:428–31. Kimura K, Mukohara N, Tsugawa C, et al. Tracheoplasty for congenital stenosis of the entire trachea. J Pediatr Surg 1982;17:869–71. Idriss FS, DeLeon SY, Ilbani MN, et al. Tracheoplasty with pericardial patch for extensive tracheal stenosis in infants and children. J Thorac Cardiovasc Surg 1984;88:527–36. Heimansohn DA, Kesler KA, Turrentine MW, et al. Anterior pericardial tracheoplasty for congenital tracheal stenosis. J Thorac Cardiovasc Surg 1991;102:710–6. Jaquiss RDB, Lusk PR, Spray TL, et al. Repair of longsegment tracheal stenosis in infancy. J Thorac Cardiovasc Surg 1995;110:1504–12. Dunham ME, Holinger LD, Backer CL, Mavroudis C. Management of severe congenital tracheal stenosis. Ann Otol Rhinol Laryngol 1994;103:351–6. Backer CL, Mavroudis C, Dunham CE, Holinger LD. Reoperation after pericardial patch tracheoplasty. J Pediatr Surg 1997;32:1108–12.
Development of Tracheal Surgery: A Historical Review
297. Tsang V, Munday A, Gilbe C, Goldstraw P. Slide tracheoplasty for congenital funnel-shaped tracheal stenosis. Ann Thorac Surg 1989;48:632–5. 298. Grillo HC. Slide tracheoplasty for long segment congenital tracheal stenosis. Ann Thorac Surg 1994;58:613–21. 299. Grillo HC, Wright CD, Vlahakes GJ, MacGillivray TE. Management of congenital tracheal stenosis by means of slide tracheoplasty or resection and reconstruction with long term follow-up of growth after slide tracheoplasty. J Thorac Cardiovasc Surg 2002;123:145–52. 300. Donahoe PK, Gee PE. Complete laryngotracheoesophageal cleft: management and repair. J Pediatr Surg 1984;19:143–8. 301. Brandenberg JH. Idiopathic subglottic stenosis. Trans Am Acad Opthalmol Otolaryngol 1972;76:1402–6. 302. Mikaelian DO. Idiopathic subglottic stenosis in an adult. J Laryngol Otol 1974;88:467–72. 303. Jazbi B, Goodwin C, Tackett D, Faulkner S. Idiopathic subglottic stenosis. Ann Otol Rhinol Laryngol 1972;86:644–8. 304. Grillo HC, Mark EJ, Mathisen DJ, Wain JC. Idiopathic laryngotracheal stenosis and its management. Ann Thorac Surg 1993;56:80–7. 305. Dedo HH, Catten MD. Idiopathic progressive subglottic stenosis: findings and treatment in 52 patients. Ann Otol Rhinol Laryngol 2001;110:305–11. 306. Ashiku SK, Kuzucu A, Grillo HC, et al. Laryngotracheal resection as an effective definitive surgical treatment for idiopathic laryngotracheal stenosis. J Thorac Cardiovasc Surg. [In press] 307. Mark JE, Patterson GA, Grillo HC. Case records of the Massachusetts General Hospital 46 – 1992. N Engl J Med 1992;327:1512–8. 308. Mark EJ, Braman SS, Grillo HC. Case records of Massachusetts General Hospital 32 – 1999. N Engl J Med 1999;341:1292–9. 309. Herzog H, Nissen R. Erschlaffung und expiratorische Invagin*tion des membranösen Teils der intrathorakalen Luftröhre und der Haupt bronchien als Ursache der asphyktischen Anfälle beim Asthma bronchiale und bei der chronischen asthmoiden Bronchitis des Lungenemphysems. Schweiz Med Wochenschr 1954;84:217–9. 310. Herzog H. Exspiratorische Stenosis der Trachea und der grossen Bronchien durch die erschlaffte Pars membranacea. Operative Korrektur durch Spanplastik. Thoraxchir 1958;5:281–319. 311. Nissen R. Tracheoplastik zur Beseitigung der Erschlaf-
312. 313. 314. 315. 316. 317.
318. 319.
320.
321. 322.
323. 324. 325.
fung des membranösen Teils der Intrathorakalen Luftröhre. Schweiz Med Wochenschr 1954;84:219–21. Rainer WG, Newby JP, Kelble DL. Long term results of tracheal support surgery for emphysema. Dis Chest 1968;53:765–72. Wright CD, Hahmoud Z, Grillo HC, Mathisen DJ. Results of membranous wall tracheoplasty for large airway expiratory collapse. [In preparation] Maier HC, Gould WJ. Agenesis of the lung with vascular compression of the tracheobronchial tree. J Pediatr 1953;43:38–42. Quillin SP, Shackleford GD. Postpneumonectomy syndrome after left lung resection. Radiology 1991;179:100–2. Grillo HC, Shepard JO, Mathisen DJ, Kanarek DJ. Postpneumonectomy syndrome: diagnosis, management and results. Ann Thorac Surg 1992;54:638–51. Shamji FM, Deslauriers J, Daniel M, et al. Postpneumonectomy syndrome with an ipsilateral aortic arch after left pneumonectomy. Ann Thorac Surg 1996;62:1627–31. Boiselle PM, Shepard JO, McLoud TC, et al. Postpneumonectomy syndrome: another twist. J Thorac Imaging 1997;12:209–11. Johnson J, Kirby CK, Lazatin CS, co*cke JA. The clinical use of a prosthesis to prevent overdistention of the remaining lung following pneumonectomy. J Thorac Surg 1949;18:164–72. Adams HD, Junod FL, Aberdeen E, Johnson J. Severe airway obstruction caused by mediastinal displacement after right pneumonectomy in a child. J Thorac Cardiovasc Surg 1972;63:534–9. Kaunitz VH, Fisher FC. Continued pneumothorax in the management of the postpneumonectomy pleural space. Ann Thorac Surg 1966;2:455–63. Powell RW, Luck SR, Raffensperger JG. Pneumonectomy in infants and children: the use of a prosthesis to prevent mediastinal shift and its complications. J Pediatr Surg 1979;14:231–7. Szarnicki R, Maurseth K, Defeval M, Stark J. Tracheal compression by the aortic arch following right pneumonectomy in infancy. Ann Thorac Surg 1978;25:231–5. Wasserman K, Jamplis RW, Lash H, et al. Postpneumonectomy syndrome: surgical correction using Silastic implants. Chest 1979;75:60–78 Rasch DK, Grover FL, Schnapf BM, et al. Right pneumonectomy syndrome in infancy treated with an expandable prosthesis. Ann Thorac Surg 1990;50:125–6.
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Part 1
DISEASES, DIAGNOSIS, RESULTS OF TREATMENT
ANATOMY, PHYSIOLOGY, PATHOLOGY, DIAGNOSTIC METHODS
DISEASES AND RESULTS OF TREATMENT
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CHAPTER ONE
Anatomy of the Trachea Hermes C. Grillo, MD
Tracheal Structure Anatomic Relationships Blood Supply Lymphatics
The trachea functions as a conduit for ventilation and also for clearance of tracheal and bronchial secretions. In disease, purulent matter and blood are evacuated via the trachea. Viewed as a simple tube, the trachea appears to be a structure ideally suited for surgical reconstruction or even for replacement following removal of a diseased segment. However, the trachea exhibits a number of anatomical features that may cause great difficulties for surgical reconstruction or replacement. These include its unpaired nature, its unique lateral semirigidity but longitudinal flexibility, its short length, its limited longitudinal elasticity, its proximity to major vascular structures, and its largely segmental blood supply. Barriers to tracheal replacement with an epithelial lined structure are discussed in Chapter 45, “Tracheal Replacement.”
Tracheal Structure The adult male trachea averages 11.8 cm in length (range 10 to 13 cm) from the lower border of the cricoid cartilage to the top of the carinal spur, varying with the patient’s height. There are usually from 18 to 22 cartilages within this length, approximating almost two rings per cm.1 Cartilaginous rings may be incomplete or bifid. The lateral tracheobronchial angles are located slightly higher than the carinal spur so that the length of the trachea proper along its lateral wall is slightly shorter than that measured anteriorly in the midline to the carinal spur (Figure 1-1). The carina in the adult projects quite consistently on the body surface at the level of the sternal angle since it is held in place by the aortic arch. The right main bronchus continues more vertically, whereas the left is always more horizontal with respect to the trachea. The angles between the bronchi and trachea vary quite widely. In infants, the subcarinal angle between the bronchi is much wider and the bronchi lie more transversely. The level of the trachea and of its lesions is often described by reference to specific vertebrae. Since levels vary in an individual with cervical flexion and extension as well as with respiration and deglutition, and among individuals by age, spinal curvature, anteroposterior diameter of the thorax, and body build, denomination by vertebral level is of little use. More to the point is the length of airway from the vocal cords to carina, or better, of the trachea from cricoid to carina, using the carinal spur as the lower point of mea-
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surement. Also important are measures of length of uninvolved trachea above and below a lesion, plus the longitudinal extent of the lesion. These distances are easily determined, although without absolute precision, by imaging (see Chapter 4, “Imaging the Larynx and Trachea”) and bronchoscopy (see Chapter 5, “Diagnostic Endoscopy”). As a practical rule, I consider about three-fifths of the juvenile trachea to reside above the sternal notch, about one-half in the young adult, and one-third or less in older adults. In the first two groups especially, the proportion of cervical trachea varies with cervical extension and flexion. In the adult, the tracheal lumen is often roughly ovoid, flattened anteroposteriorly (Figure 1-2). The rings are normally C-shaped, with the posterior membranous wall connecting the arms of the “C” in an essentially straight line measuring generally less than one-third of the circumference of the trachea (see Figure 1-2A–C). The proportion of cross-sectional cartilaginous length to length of membranous wall does not change from infancy during growth.2 An adult tracheal ring is about 4 mm high. Therefore, there are about two rings per cm of trachea. The length and diameter of the trachea is roughly proportional to the size of the individual. Men generally have a trachea of larger diameter than women (see Figures 1-2B,C). In the adult male, the external diameters of the trachea measure about 2.3 cm coronally and 1.8 cm sagitally. Corresponding figures in the female are 2.0 and 1.4 cm. The tracheal wall is about 3 mm thick. The trachea narrows somewhat as it progresses distally to the carina, more notably in children. These are important points in selecting an endotracheal tube, especially for ventilation, where the tube may be in place for a long time. A small man or woman, even if obese, will nonetheless have a trachea of shorter length and narrower diameter. An excessively wide tube can produce subglottic erosion and consequent stenosis. Griscom and Wohl together with Fenton measured the length, diameters, cross-sectional area, and volume of the trachea in children under the age of 6 years, asleep or resting quietly, and of older children and adolescents at total lung capacity using computed tomography (CT) scans (Table 1-1).3–5 All parameters correlated with body height with little variance for gender in young children. Considerable variation in measurements of length and diameter are recorded in the literature depending upon the artifacts of handling specimens, the methods of observation, and whether the observations were made in the living or postmortem. The techniques involved measurements of fixed and unfixed autopsy specimens, the use of x-ray or CT images, and bronchoscopy. In small children, body weight may actually correlate better with tracheal growth than height or age. Increase in length outstrips growth of cross-sectional area in the first year of life.6 Thereafter, the rate of lengthening falls below the rate of area growth until puberty.2 Initially, the anteroposterior diameter is slightly greater than the transverse, producing a nearly circular lumen (see Figure 1-2A). Gradually, as the child grows, the adult configuration emerges. At first, the trachea is somewhat funnel-shaped, but the discrepancy between the area at the subcricoid level and the carina gradually diminishes, first to a cylindrical form and later to the more ovoid adult shape (see Figure 1-2B).2,6 After age 14 years, female tracheae generally stop growing whereas male tracheae continue to enlarge in cross section but not in length.4 Great care must be taken not to use excessively large endotracheal tubes for ventilation in infants and children. Formulas for selecting tube size with relation to age are not of great value because of individual variation. The shape of the adult trachea varies even without disease. Some remain nearly circular rather than becoming ovoid (see Figure 1-2C). In others, the sagittal diameter may be greater than the coronal. A slightly triangular configuration occurs less often. Unique and unexplained distortions also occur. The crosssectional area as well as shape changes dynamically with intraluminal pressure alterations due to cough (see Figure 1-2B), respiration, and ventilation; tracheal length and volume vary similarly. The aorta may displace and deform the lower trachea (see Figure 1-2D). The cross-sectional configuration of the trachea may be markedly altered with increasing age, particularly in the presence of chronic obstructive lung disease. The lower two-thirds of the trachea may gradually become flattened from side to side with a consequent decrease in lateral diameter and increase in anteroposterior diameter (see Figure 1-2E). This deformation is called, “saber sheath trachea.”7 In this
Anatomy of the Trachea
Lesser cornu Hyoid bone Greater cornu Thyrohyoid membrane Superior cornu Superior thyroid notch Thyroid cartilage Anterior thyroid lamina Inferior cornu Cricoid cartilage Cricothyroid membrane
Carina
RMB
LMB
FIGURE 1-1 Principal features of the larynx and trachea. Anterior view. Tracheobronchial angles vary widely. LMB, RMB = left, right main bronchus.
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A
E
B
F
C
G
D
H
1-2 Cross-sectional configuration of the trachea in health and disease. A, Juvenile trachea appears quite circular. B, Adult male trachea at rest on the left. The shape of the lumen can also be more circular. With cough, the membranous wall “accordions,” pulling cartilage together (at the right). C, Female trachea is smaller in diameter. In this case, the lumen is circular. D, Common deformity related to proximity to the aorta just above the left main bronchus. E, Saber-sheath trachea. The walls are not malacic. F, Above. Triangular deformation which occurs in some patients with chronic obstructive pulmonary disease, especially in proximal trachea. Middle. In the thoracic trachea, flattening and softening of cartilage occurs, with elongation of membranous wall. Below. Obstructive approximation of anterior and posterior walls with expiratory effort or cough. G, Tracheopathia osteoplastica. Saber-sheath configuration with characteristic submucosal osseocartilaginous nodules. H, Deformity occurring in tracheobronchomegaly, Mounier-Kuhn disease. The initially huge circular lumen may become distorted, as shown. The cartilage remains firm even when angulated. FIGURE
case, the rings are not malacic and indeed may even calcify. Rarely, obstruction follows if the deformity narrows the airway markedly. Another deformation in chronic obstructive pulmonary disease is anteroposterior flattening of the thoracic trachea, which is accompanied by softening of the rings (see Figure 12F). The cartilages may assume the configuration of an archer’s bow. The membranous wall widens and becomes redundant. These changes in configuration and accompanying malacia result in various degrees of tracheal obstruction, notably in expiration and on cough (see Figure 1-2F). The upper trachea may become triangular in these patients (see Figure 1-2F). Unusual tracheal configurations occur in unique tracheal diseases such as tracheopathia osteoplastica (see Figure 1-2G) and tracheobronchomegaly (see Figure 1-2H). With advancing age or as a result of local trauma or disease, the tracheal rings and laryngeal cartilages may calcify. The only complete cartilaginous ring in the normal airway is the cricoid cartilage. Congenital tracheal stenosis (see Chapter 6, “Congenital and Acquired Tracheal Lesions in Children”) is characterized by segments of variable length composed of completely circular rings of cartilage or, rarely, by irregular circular plates. The cricoid level is the narrowest point in the upper airway below the glottis both in children and in adults. The cricoid has a broad posterior plate and is shaped much like a reversed signet ring (Figures 1-3A,B). The first ring of the trachea may be wider and partly recessed into the lower margin of the cricoid. The endoscopist must appreciate that the vocal cords lie approximately in midlarynx, just above the level of the anterior inferior margin of the thyroid cartilage, and that there is about 1.5 cm of subglottic larynx present between the vocal cords and the lower margin of cricoid (see Figures 1-3A–D). Only there does
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Anatomy of the Trachea
the trachea actually begin. Endoscopists sometimes underestimate the involvement of subglottic larynx by a pathologic process, believing they are examining the trachea once they pass the vocal cords. The conus elasticus is a dome-shaped configuration just below the vocal cords (see Figure 1-3D). Laryngeal anatomy is briefly described in Chapter 35, “Laryngologic Problems Related to Tracheal Surgery.” Particularly in youth, the tracheal wall is quite elastic laterally. The normally thin cartilages in childhood are more easily compressed than those of the young adult, and these more easily than cartilages in the older adult. With normal cough, the membranous wall accordions, with the trachealis muscle pulling the lateral cartilaginous walls of the trachea medially (see Figure 1-2B). The intercartilaginous muscles between the rings and muscles in the membranous wall contract simultaneously. The tracheal walls may also be deformed by lateral pressure of the aortic arch (see Figure 1-2D), the brachiocephalic artery, and by extrinsic masses (see Chapter 15, “Tracheobronchial Malacia and Compression”). The trachea also bends flexibly but there is limited longitudinal extensibility, somewhat greater in the young. It is estimated to amount to about 10%. Flexibility and elasticity become more limited with advancing age, particularly when calcification occurs in cartilages. The mucosa of the trachea is tightly applied to the inner surface of the cartilage. The two are not easily dissected apart. Mucosa covers the posterior muscular membranous wall as well. The mucosa is normally respiratory in character, being ciliated pseudostratified columnar epithelium. Goblet cells are liberally present. Submucosal mucous glands are numerous and connect to the surface by ducts. In habitual smokers and others with chronic irritation, squamous metaplasia may occur and the cilia are destroyed. Secretions may be successfully cleared by cough despite metaplasia and even when the mucosa has been totally replaced by either a cutaneous tube reconstruction or by interposition of synthetic materials.
Anatomic Relationships The normal trachea viewed anteroposteriorly lies in the midline, connecting the larynx with the carina. In the lateral view, however, the trachea is tilted, slanting from an anterior, nearly subcutaneous position just below the larynx to a posterior one at the carinal level, where it lies against the esophagus close to the vertebrae. Although there is great individual variation, this angle from the vertical gradually increases with age
Table 1-1 Tracheal Dimensions in Children and Young Adults Internal Diameters
Cross-sectional
Age (Years)
Height Percentile
Tracheal Length (cm)
Anteroposterior (cm)
Transverse (cm)
Area (cm2)
Volume (cm3)
0–2 2–4 4–6 6–8 8–10 10–12 12–14
40 47 57 54 54 49 58
5.4 6.4 7.2 8.2 8.8 10.0 10.8
0.53 0.74 0.80 0.92 1.03 1.16 1.30
0.64 0.81 0.90 0.93 1.07 1.18 1.33
0.28 0.48 0.58 0.69 0.89 1.10 1.39
1.57 3.11 4.16 5.67 7.87 11.1 15.4
14–16 16–18 18–20
F 67 47 60
M 53 47 62
F 11.2 12.2 11.8
M 12.4 12.4 13.1
F 1.39 1.37 1.42
M 1.45 1.57 1.75
F 1.46 1.40 1.39
M 1.43 1.59 1.66
F 1.62 1.54 1.59
M 1.62 2.01 2.30
F 18.2 18.8 18.9
Data from computed tomography measurements in life, near total lung capacity if over 6 years.4 “Tracheal length” is from vocal cords to carina. F = female; M = male.
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Epiglottis Supraglottic larynx
Glottis (true vocal cords) Subglottic larynx Vocal cords
Recurrent laryngeal nerve
A
B
Preepiglottic space
Epiglottis Ventricular fold (false cord)
Hyoid bone
Supraglottic larynx
Ventricle Anterior lamina thyroid carilage
Glottis (true vocal cords) Arytenoid muscle
Vocal cord Cricoid
Subglottic larynx
Cricoid
C
D
FIGURE 1-3 External laryngeal relationships. Anterior (A) and lateral (B) views. Note the position of the true vocal cords (vocal folds) in the midlarynx. The cricoid cartilage shows the configuration of a reversed signet ring. The inferior cornu of the thyroid cartilage is close to the entry point of the inferior laryngeal nerve. C, Lateral view of the interior of the larynx. Anterior surface to the left. Note the relationships of the ventricular fold (false vocal cord), ventricle and vocal fold (true vocal cord), and their locations. The subglottic larynx lies between the glottis and inferior cricoid border. D, Diagram of interior configuration of larynx (anterior view). Note the dome-shaped airway beneath the glottis. This is the conus elasticus, shaped by intrinsic muscles. See Chapter 35, “Laryngologic Problems Related to Tracheal Surgery,” for description of intrinsic laryngeal musculature.
Anatomy of the Trachea
(Figure 1-4). In many old people, the trachea becomes increasingly horizontal in its course from the larynx to the carina and may approach a nearly transverse position. This worsens with severe kyphosis. The sternum also tends to flare out with aging. The larynx lies closer to the sternal notch with increasing age and the trachea loses mobility upon attempted cervical extension. This explains how subglottic damage may be occasioned by upward and backward erosion of a tracheostomy tube, even though the stoma was placed at a correct level in the trachea. In youth, a large proportion of the trachea presents in the neck above the level of the sternal notch even when the neck is in neutral position. With extension, more than half of the trachea rises into the neck, and sometimes, by as much as two-thirds (see Figure 1-4A). In contrast, attempted cervical extension in old age may bring very little, if any, trachea into the neck (see Figure 1-4B). The surgical implications are clear. The amount of trachea that can be brought into the neck on hyperextension of the cervical spine determines the percentage of trachea that may be resected and approximation obtained by cervical flexion alone.8
A
a
Neutral position
b
Extension
c
Flexion
B
a
b
c
FIGURE 1-4 Tracheal position in youth and old age with cervical extension and flexion. The trachea is much more vertical on lateral projection in youth (A) than in old age (B). A, (a) In youth, approximately one-third of the trachea is in the neck above the sternum (dashed line) in neutral position. (b) With cervical extension, one-half or more rises into the neck. (c) Most of the trachea devolves into the thorax on full flexion. B, In the aged, the level of the larynx (a) changes little with attempted cervical (b) extension and (c) flexion. Surgical implications are obvious.
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The esophagus lies in close relation to the trachea throughout its course (Figure 1-5). The esophagus commences at the level of the posterior cricoid, attached to it by the sling of cricopharyngeus muscle. Since the esophagus is a little to the left, the right posterior margin of the trachea is immediately in front of the vertebral bodies. In inflammatory disease, this portion of the posterior tracheal wall can adhere to the vertebral bodies. A layer of areolar tissue lies between the membranous wall of the trachea and the esophagus. This close juxtaposition of the walls of these two organs has been termed the “party wall.” Normally, the plane is easily separable. A common blood supply, as noted below, is shared by these two tubular organs. Anteriorly, the thyroid isthmus usually crosses and is closely applied to the trachea at the level of the second and third rings (see Figure 1-5A). The isthmus is sometimes very broad, but in a very few patients is absent. The pyramidal lobe commonly arises from the isthmus, often slightly to the left. The lateral lobes of the thyroid gland are also closely applied to the anterolateral and lateral walls of the trachea. Multiple small blood vessels, lymphatic channels, and fibrous attachments bind the isthmus and adjacent portions of the thyroid lobes to the tracheal wall.9 The inferior thyroid artery supplies the lower portion of the thyroid gland and contributes importantly to the blood supply of the upper trachea. Details are provided below. The superior laryngeal nerves concern the tracheal surgeon in connection with laryngeal release procedures and thyroidectomy. An external branch lies deep and parallel to the superior laryngeal artery and innervates the cricothyroid muscle. It gives a branch to the inferior pharyngeal constrictor. The internal branch passes into the thyrohyoid membrane with the superior laryngeal artery. It provides sensation to laryngeal mucosa and hence reflex protection to the larynx.10 The recurrent laryngeal nerves follow different courses right and left (see Figure 1-5). The left nerve originates from the vagus beneath the arch of the aorta and lies close to the tracheoesophageal groove along its entire course. The right nerve loops around the subclavian artery and therefore approaches the tracheoesophageal groove from a more lateral position. The right recurrent laryngeal nerve often passes between branches of the right inferior thyroid artery whereas the left often is posterior to the left inferior thyroid artery.10 They enter the larynx between the cricoid and thyroid cartilages deep to the inferior cornua of the thyroid cartilage, behind the articulation of the thyroid and cricoid cartilages, to innervate the intrinsic laryngeal muscles.10–12 Small branches travel to the trachea, trachealis muscle, esophagus, and inferior constrictors, including the cricopharyngeus muscle. Proximal branches near the recurrent nerve loops lying beneath the right subclavian artery and aorta on the left contribute to the cardiac plexus intrathoracically.10 Rarely, the right inferior laryngeal nerve is not recurrent but crosses the neck transversely from the vagus in one or more branches to enter the larynx. This occurs in conjunction with an anomalous right subclavian artery arising from a left aortic arch and passing posterior to the esophagus. The nonrecurrent nerve passes from the vagus beneath the carotid artery, may have two terminal branches, and may also give off branches to the trachea, esophagus, and thyroid. Even more rarely, the left inferior laryngeal nerve may be nonrecurrent in conjunction with the right aortic arch and left retroesophageal aberrant subclavian artery. Estimated incidence is 0.63% on the right and 0.04% on the left.13 The left brachiocephalic vein is well anterior to the pretracheal plane. The brachiocephalic artery, however, crosses over the midtrachea obliquely from its point of origin from the aortic arch to reach the right side of the neck (see Figures 1-5 through 1-7). In children, the artery rises higher and is encountered in the lower part of the extended neck. In young adults also, this artery crosses the trachea at the base of the neck with even moderate cervical extension. Thus in the young, a large proportion of the trachea and the brachiocephalic artery regularly rise into the neck on extension (see Figure 1-4). If a tracheostomy is placed in a child or young adult with reference to the sternal notch rather than the cricoid cartilage, it is easy to see how tracheal arterial fistula can occur (see Chapter 13, “Tracheal Fistula to Brachiocephalic Artery”). The brachiocephalic artery branches into the right common carotid and subclavian arteries a short distance to the right of the trachea and behind the origin of the internal jugular vein. The left common carotid nor-
Anatomy of the Trachea
mally arises a short distance from the origin of the brachiocephalic artery from the aorta forming a “V” slightly to the left of the tracheal midline. The brachiocephalic artery and the left common carotid artery may arise from a common arterial trunk, which overlies the trachea (see Figure 1-7).14 Anatomic variation can be important if the trachea is adherent to the back of a common trunk (see Figure 1-7) as a result of inflammation or prior tracheal surgery. Occasionally (less than 10%), a small thyroidea ima artery arises from the back of the brachiocephalic artery and travels superiorly to the thyroid gland. At the carinal level, the left main bronchus passes beneath the aortic arch and the right main bronchus beneath the azygos vein. The superior vena cava lies just anterior and to the right of the trachea. The pulmonary artery lies inferiorly in front of the carina (see Figures 1-5, 1-6). Thus, in anterior approach to the carina, a deep quadrilateral space is developed transpericardially in front of the carina, bordered by the
Right vagus nerve Left common carotid artery Right subclavian artery Left subclavian artery Right recurrent laryngeal nerve
Right common carotid artery Left vagus nerve
Right and left brachiocephalic veins
Aorta
Brachiocephalic artery
Superior vena cava
Left recurrent laryngeal nerve
Pulmonary trunk
A
FIGURE 1-5 Relationships of trachea to surrounding structures. A, Anterior view. Note the tight packing of major mediastinal vessels adjacent to the trachea.
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Vagus nerve Inferior cornu of thyroid cartilage
Cricoid cartilage
Right carotid artery
Thyroid gland Brachiocephalic artery
Right subclavian artery
Arch of aorta Right recurrent laryngeal nerve
Trachea
1 Azygos vein
Carina 2 RMB Inferior vena cava
LMB
Phrenic nerve Pulmonary veins B
Pulmonary artery
FIGURE 1-5 (CONTINUED) B, Right-sided oblique view shows the complete access to the intrathoracic trachea, crossed only by the vagus nerve and azygos vein. LMB, RMB = left, right main bronchus.
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49
Thymus
Right brachiocephlic vein
Left brachiocephalic vein Phrenic nerve
Phrenic nerve
Brachiocephalic artery Left common carotid artery Vagus nerve
Trachea
Left subclavian artery
Vagus nerve
Recurrent laryngeal nerve Esophagus
A
Thoracic duct
T3-4
Thymus Phrenic nerve
T3-4
A
T4-5
B
Phrenic nerve
Superior vena cava
Aortic arch
Azygous vein Trachea
Vagus nerve
Vagus nerve
Recurrent laryngeal nerve
Esophagus Thoracic duct
B
T4-5
1-6 Cross-sectional computed tomography views of tracheal anatomic relationships in the mediastinum. Diagram shows level of sections A at T3-4, and B at T4-5. A, Thoracic trachea. B, Supracarinal trachea. Mediastinal structures are labelled.
FIGURE
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A
B
C
D
FIGURE 1-7 Many variations occur in the arrangement of the branches arising from the aortic arch. The two most common patterns are (A) with separate origins of brachiocephalic and left carotid arteries, and (B) with a common origin. In both, a separate left vertebral artery may arise from the arch distal to the left carotid (C,D) and confuse the tracheal surgeon. The common origin may complicate resection of tracheal lesions that are adherent to the trunk or the treatment of tracheoarterial fistula. Adapted from Williams GD and Edmonds HW.14
Anatomy of the Trachea
superior vena cava on the right, the aortic arch on the left, the pulmonary artery inferiorly, and the brachiocephalic vessels superiorly (see Figure 23-7 in Chapter 23, “Surgical Approaches”). The presence of major vascular structures in close proximity to the trachea makes exposure of its full extent difficult through any single incision. These anatomic facts must be considered in planning surgical approach to a tracheal lesion (see Chapter 23 “Surgical Approaches”). The previously undisturbed pretracheal plane, except for the point of attachment of the thyroid isthmus, may be easily developed bluntly because it consists of areolar tissue with few blood vessels. Normally, it is essentially avascular except for the rare thyroidea ima artery or an even rarer small posterior branch from the brachiocephalic artery to the trachea. A few inferior thyroid veins overlie the upper trachea immediately below the thyroid isthmus. These drain into the left brachiocephalic vein most commonly. The attachments of connective tissue to the trachea are loose enough so that vertical movement is easily possible to a considerable degree both functionally and surgically. The trachea is, however, fixed by the sling of the aortic arch over the left main bronchus where relatively little sliding motion occurs. With the increasing anteroposterior diameter of the thorax with age, related to vertebral kyphosis, this point of fixation draws the carina further posteriorly and the trachea falls into a more horizontal position when viewed laterally (see Figure 1-4B). Mobility of the trachea with cervical extension becomes limited, as previously noted.
Blood Supply Prior to the development of tracheal surgery, detailed description of the arterial blood supply of the trachea was unknown. Using radiographs of injected specimens of the human trachea, Miura and Grillo showed that blood supply of the cervical trachea originates from the inferior thyroid artery in a variable pattern (Figure 1-8).15 The blood supply enters the trachea through lateral tissue pedicles in segmental fashion throughout the trachea. Complete description of the entire tracheal blood supply was made by Salassa and colleagues (Figure 1-9).16 The upper half of the trachea is supplied in most cases by three tracheoesophageal branches of the inferior thyroid artery (see Figure 1-8A). The first branch supplies the lower cervical trachea with no or minor contributions to the esophagus. The second supplies the middle section of the cervical trachea, and the third the upper section. Both of these branches contribute to esophageal blood supply. The tracheal branches pass either anterior or posterior to the recurrent laryngeal nerves or both. The pattern varies, and there may be only one or two arteries. One or other artery may predominate (see Figure 1-8B). In 2 of 17 specimens studied, the lower cervical trachea was supplied instead by a branch originating from the subclavian artery (see Figures 1-8Ad,Ae). The superior thyroid artery does not give direct branches to the trachea, but it does anastomose with the inferior thyroid artery and also contributes with fine branches running from the thyroid isthmus to the adjacent tracheal wall. The bronchial arteries provide consistent blood supply to the carina and lowermost trachea (see Figure 1-9).16 An anterior branch of the superior bronchial artery originates from the right side of the aorta posteriorly. This branch usually travels over to the proximal left main bronchus to the anterior carina. The principal and posterior branches of this vessel pass behind the esophagus to the right main bronchus. One of these branches may arise from a supreme intercostal artery. The middle bronchial artery courses around the medial aspect of the left bronchus and anastomoses at the carina with the superior bronchial artery or higher tracheal vessels. The inferior bronchial artery appears to supply chiefly the left bronchial tree. The left main bronchus is most often supplied by two left-sided aortic branches. Bronchial artery patterns are very varied (Figure 1-10).17 Flow in bronchial arteries after main bronchial artery division is given detailed consideration in Chapter 44, “Airway Management in Lung Transplantation.” These considerations apply in carinal resection and reconstruction, although not as critically as in lung transplantation.
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a
A
b 32%
50%
d 7%
c 7%
e 4%
FIGURE 1-8 A, Variants in distribution of inferior thyroid artery to cervical trachea. Distribution in 28 specimens. The frequency of each pattern is indicated. In (d) and (e), the major vessel is the subclavian artery.
Anatomy of the Trachea
a 68%
b 14%
c 4%
d 14%
B 1-8 (CONTINUED) B, Frequency of predominance of inferior thyroid artery branches. Adapted from Miura T and Grillo HC.15
FIGURE
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Superior thyroid artery
Inferior thyroid artery Vertebral artery
Costocervical trunk 3rd branch
Supreme intercostal artery
2nd branch 1st branch Internal thoracic artery
Lateral longitudinal anastomosis
Superior bronchial artery Middle bronchial artery
A FIGURE 1-9 Tracheal blood supply. A, Left anterior view. Reproduced, by permission of Mayo Foundation for Medical Education and Research, from Salassa JR et al.16
The balance of the middle and lower tracheal blood supply is derived in variable fashion from a brachiocephalic-subclavian system: from the supreme intercostal artery, the subclavian artery, the right internal thoracic artery, and the brachiocephalic artery (see Figure 1-9).
Anatomy of the Trachea
Inferior thyroid artery Vertebral artery Innominate artery
Subclavian artery
Supreme intercostal artery
2nd branch 1st branch
Internal thoracic artery
Aorta
Esophagus
B FIGURE 1-9 (CONTINUED) B, Right anterior view. Note the basically segmental nature of distribution. Reproduced, by permission of Mayo Foundation for Medical Education and Research, from Salassa JR et al.16
Salassa and colleagues identified from three to seven principal tracheal arteries along the entire length of the lateral tissue pedicles.16 Just lateral to the tracheoesophageal groove, the primary vessels divide into tracheal and esophageal branches (Figure 1-11). The tracheal branches pass directly to the tracheal wall,
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A 40.6%
C 20.6%
B 21.3%
D 9.7%
1-10 Principal patterns of bronchial artery supply to the trachea and bronchi. Frequency of occurrence is noted. These patterns account for over 92% of variations. Not shown in these diagrams are the proximal branch of the superior bronchial artery which courses anteriorly over the left main bronchus to carina (see Figure 1-9A) and the middle bronchial branch passing beneath the left main bronchus to carinal anastomosis (see Figure 1-9B). Adapted from Cauldwell EW et al.17
FIGURE
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branching up and down over the width of several rings. These fine branches in turn connect with the branches of the next segmental vessels above and below. These vessels form a somewhat irregular but generally complete series of fine longitudinal anastomoses on the wall of the trachea. From the vessels that reach the trachea, transverse intercartilaginous arteries extend deeply into the tracheal wall and anastomose with those from the opposite side at the midline (see Figure 1-11). These vessels branch into the submucosa. Smaller intercartilaginous branches point posteriorly and terminate in the membranous tracheal wall. The posterior membranous wall of the trachea is also supplied by secondary small branches from the primary esophageal vessels branching from the tracheoesophageal arteries. Welldeveloped longitudinal anastomoses are also present. The tracheal cartilages receive nourishment from the submucosal plexus only. The submucosal plexus of both the mucosa overlying the cartilages and that overlying the membranous wall interconnect and are important in supplying the membranous wall. Although a large part of the length of the trachea can usually be circumferentially dissected without necrosis if the trachea remains intact (and with it the vertical longitudinal vessels), circumferential dissec-
Coronal section of tracheal wall… Anterior transverse intercartilaginous artery
Lumen
Trachea
Submucosal capillary plexus Transverse intercartilaginous artery
Lateral longitudinal anastomosis
Primary tracheal artery Posterior transverse intercartilaginous artery Pattern of microvasculature of mucosa Tracheoesophageal artery Primary esophageal artery
Esophagus
Muscular posterior wall of trachea
Secondary tracheal twig to posterior wall
FIGURE 1-11 Microscopic blood supply of the trachea. See text. Reproduced, by permission of Mayo Foundation for Medical Education and Research, from Salassa JR et al.16
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tion of an excessively long segment of trachea above or below a point of tracheal division can lead to devascularization. Necrosis may follow. No absolute distances acceptable for toleration of circumferential dissection have been experimentally determined since the intimate blood supply of the trachea varies with species. Clinical experience, however, dictates the wisdom of minimizing circumferential dissection of trachea that is to remain in the patient, with a goal of dissecting free no more than 1 or 2 cm of trachea above or below an anastomotic line. Mediastinal lymph node dissection for tracheal tumors should be limited to lymph nodes immediately adjacent to the segment to be resected, in order to avoid contributing to devascularization. Surgical caution applies to the very rare situation where circumferential tracheal resection may seem to be desirable during concomitant esophagectomy. Tracheal necrosis may well follow because the tracheoesophageal arteries are interrupted by esophagectomy.18
Lymphatics Detailed studies of tracheal lymphatic drainage are few. Following submucosal injections of India ink and dye into the canine trachea, Strauss observed that 1) 2) 3) 4) 5) 6) 7) 8)
tracheal lymphatic vessels were present as fine intercellular spaces beneath the mucous membrane over the cartilages, the lymph flowed up or down the trachea to the nearest interspace between cartilages, one to three trunks flowed horizontally in the interspaces between rings, flow from the anterior wall went to either side, flow from the lateral wall passed to the membranous wall, the membranous wall contained larger vessels in greater numbers and particles traveled up and down the wall, there were more horizontal collecting vessels in the lower interspaces, especially near the carina, and lymph vessels left the tracheal wall, especially at the lower end of the trachea, and passed to perivascular lymphatics and then to lymph nodes along the trachea.19
Primary tracheal lymph nodes are pretracheal, paratracheal, and subcarinal.20 The anatomy of the mediastinal lymph nodes has been well described in connection with lung cancer in many places.21 Pathways of lymph drainage from nodes along the trachea have been elucidated somewhat by Ricquet and colleagues.22 The right lower paratracheal lymph nodes drain into thoracic duct tributaries which travel along the course of the azygos vein. Left superior bronchial nodes below the trachea drain directly to the mediastinal thoracic duct or to the arch of the duct via the left recurrent chain. An alternative pathway is to the aortic arch node and up along the arch. Tracheal bifurcation nodes drain through accessory ducts on either side of the esophagus to the mediastinal thoracic duct. Clinical observations of peritracheal, pretracheal, and subcarinal nodal metastases, principally from primary squamous and adenoid cystic carcinomas of the trachea, not surprisingly show that cells reach the nodes most often nearest to the tumor. I have not often seen obvious “skip” nodal involvement. Although limited node dissection has been practiced in order to preserve tracheal blood supply, recurrence in most cases has been distant rather than local. It must, however, be noted that mediastinal irradiation has been given quite routinely postoperatively. These are admittedly anecdotal observations. A limited number of intratracheal dye injections in vivo by Miura and Grillo (unpublished, 1965) confirmed tracking to the nearest paratracheal mediastinal lymph nodes. I believe that primary tracheal tumors with mediastinal lymph node involvement should be thought of as N1 disease.
Anatomy of the Trachea
References 1.
2. 3. 4. 5. 6. 7. 8. 9.
10. 11. 12.
Grillo HC, Dignan EF, Miura T. Extensive resection and reconstruction of mediastinal trachea without prosthesis or graft: an anatomical study in man. J Thorac Cardiovasc Surg 1964;48:741–9. Wailoo MP, Emery JL. Normal growth and development of the trachea. Thorax 1982;37:584–7. Griscom NT, Wohl MEB. Dimensions of the growing trachea related to body height. Am Rev Respir Dis 1985;131:840–4. Griscom NT, Wohl MEB. Dimensions of the growing trachea related to age and gender. Am J Roentgenol 1986;146:233–7. Griscom NT, Wohl MEB, Fenton T. Dimensions of the trachea to age 6 years related to height. Pediatr Pulmonol 1989;6:186–90. Butz RO. Length and cross section growth pattern in the human trachea. Pediatrics 1968;42:336–41. Greene RE, Lechner GL. “Saber-sheath” trachea. A clinical and functional study of marked coronal narrowing of the intrathoracic trachea. Radiologe 1975;115:265–8. Mulliken J, Grillo HC. The limits of tracheal resection with primary anastomosis: further anatomical studies in man. J Thorac Cardiovasc Surg 1968;55:418–21. Shin DH, Mark EJ, Suen HC, Grillo HC. Pathological staging of papillary carcinoma of the thyroid with airway invasion based upon the anatomic manner of extension to the trachea. Hum Pathol 1993; 24:866–70. Monfared A, Corti G, Kim D. Microsurgical anatomy of the laryngeal nerves as related to thyroid surgery. Laryngoscope 2002;112:386–92. Wang CA. The use of inferior cornu of the thyroid cartilage in identifying the recurrent laryngeal nerve. Surg Gynecol Obstet 1975;140:91–4. Pearson FG, Cooper JD, Nelems JM, Van Nostrand AWP.
13.
14.
15. 16. 17. 18. 19. 20. 21.
22.
Primary tracheal anastomosis after resection of the cricoid cartilage with preservation of recurrent laryngeal nerves. J Thorac Cardiovasc Surg 1975;70:806–16. Henry J-F, Audiffret J, Denizot A, Plan M. The nonrecurrent inferior laryngeal nerve: review of 33 cases, including two on the left side. Surgery 1988; 104:977–84. Williams GD, Edmonds HW. Variations in the arrangement of the branches arising from the aortic arch in American whites and negroes (a second study). Anat Rec 1935;62:139–46. Miura T, Grillo HC. The contribution of the inferior thyroid artery to the blood supply of the human trachea. Surg Gynecol Obstet 1966;123:99–102. Salassa JR, Pearson BW, Payne WS. Gross and microscopical blood supply of the trachea. Ann Thorac Surg 1977;24:100–7. Cauldwell EW, Sickert RG, Lininger RS, Anson BJ. The bronchial arteries. An anatomic study of 150 human cadavers. Surg Gynecol Obstet 1948; 86:395–412. Fujita H, Kawahara H, Hidaka M, et al. An experimental study on viability of the devascularized trachea. Jpn J Surg 1988;18:77–83. Strauss JF. The intimate lymphatics of the trachea. Ann Otol Rhinol Laryngol 1922;31:715–26. Rouvière H. Anatomy of the human lymphatic system. Tobian MJ, translator. Ann Arbor (MI): Edwards Bros; 1938. Shields TW. Lymphatics of the lungs. In: Shields TW, LoCicero J, Ponn RB, editors. General thoracic surgery. 5th ed. Philadelphia: Lippincott Williams & Wilkins; 2000. p. 77–90. Ricquet M, Le Pimpec Barthes F, Souilamas R, Hidden G. Thoracic duct tributaries from intrathoracic organs. Ann Thorac Surg 2002;73:892–9.
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C H A P T E R T WO
Physiology of the Trachea David J. Kanarek, MD
Anatomy Flow Volume Loop Upper Airway Obstruction
Anatomy Some knowledge of tracheal anatomy is relevant to appreciating the physiological characteristics of the trachea. The trachea is a tube of approximately 11 cm in length and 1.6 to 2.4 cm in width. It is composed of C-shaped cartilage rings, spanned by the trachealis muscle, which provides support anteriorly and laterally, and a posterior membrane that stretches and can become partially redundant when exposed to high extrapleural pressures. The distal two-thirds of the trachea is intrathoracic, whereas the proximal third is extrathoracic when the neck is extended and intrathoracic with the neck flexed. Thus, there is a zone of the trachea that can be exposed to either intrapleural or extrapleural pressure.
Flow Volume Loop The flow volume loop (FVL) has become the standard method for assessing the volume and flow characteristics of the lung, as well as for physiologic evaluation of the upper airway (Figure 2-1). The total volume exhaled, the forced vital capacity (FVC), is plotted against the simultaneously obtained flow rate (see Figure 2-1A). Flow is obtained either directly from a pneumotachograph or by the derivative of volume against time (flow). Spirometry displays volume against time, and although it yields similar information on the expiratory curve, it has less of a pictorial element. The inspiratory portion of the FVL is important in diagnosing nonfixed or variable forms of upper airway obstruction, and it is not represented in spirometric tracings. Understanding the influence of pressure changes with phases of respiration is essential in following the methods of diagnosis of upper airway obstruction. The key factor is the transluminal pressure, which is the relationship between intraluminal and extraluminal pressures. The extraluminal pressure is atmospheric pressure in the extrathoracic region and intrapleural pressure in the intrathoracic area. With inspiration, the diaphragm and chest wall muscles expand the thoracic cavity, decompressing the intrathoracic gases. This causes a negative pressure within the pleura that is greater than the negative intraluminal pressure, thus dilating the intrathoracic airways. The positive extrathoracic extraluminal pressure does not col-
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A
B
C
D
2-1 Diagrammatic representation of a normal flow volume loop (A) and different types of upper airway obstruction (B–D). Flow is the vertical axis and volume is the horizontal axis. MEF50 and MIF50 are the maximum expiratory and inspiratory flows, respectively, at 50% vital capacity.
FIGURE
lapse the extrathoracic trachea because of the rigidity of the trachea and stiffening of pharyngeal muscles by neurological influences. During unforced exhalation, the intraluminal airway pressure becomes positive relative to both the intrapleural and extrathoracic atmospheric pressures. With forced exhalation, however, the intrapleural pressure may exceed the intraluminal pressure and cause dynamic compression of the airway. This becomes important in interpreting the FVL, and in diagnosing the site and nature of an obstruction. The normal expiratory FVL has two components (see Figure 2-1A). The first part is approximately the initial 20% of the FVC, comprised of the rise and the initial descent of flow rate, and is effort dependent. The remainder is effort independent, meaning that, above a moderate effort, no increase in flow is obtained by increasing muscular effort. This is related to the phenomenon of dynamic compression, in which high intrapleural pressure, caused by contraction of the diaphragm and intercostal muscles, is added to the elastic recoil of the distended alveoli to compose the intra-alveolar driving pressure. However, the same intrapleural pressure also compresses the airways, leaving alveolar elastic recoil as the sole driving pressure, irrespective of the degree of muscular effort. At high lung volume, airflow increases with effort because high intrapleural pressures cannot be developed and because elastic recoil is high. Furthermore,
Physiology of the Trachea
the so-called choke point, the point at which intraluminal pressures equal extraluminal pressures, is in the trachea, and the more proximal trachea with its cartilaginous support is resistant to collapse. This choke point moves more distally with decreasing volume to a point where the airways are susceptible to pressure and collapse. The expiratory portion of the FVL of emphysema (Figure 2-2) has a recognizably different shape from a normal FVL. The resistance of the small airways is increased by intrinsic disease and by the loss of the tethering effect of alveoli on the airways. In addition, there is loss of alveolar elastic recoil as a result of alveolar destruction. Thus, at every lung volume, flow rates are lower than normal. In addition, the expiratory loop becomes concave downwards. The maximum flow of the inspiratory loop may be almost normal because the laxity of the airways allows them to be easily opened by negative intrapleural pressure, or the flow may be reduced when intrinsic inflammatory disease stiffens airways, producing increased resistance to flow. Proper performance of the FVL is extremely important in interpretation (Figure 2-3). A poor initial expiratory effort may produce an apparent plateau on the expiratory loop and simulate a variable intrathoracic obstruction such as tracheomalacia. This often presents as a rounding rather than a plateau-like flat-
FIGURE 2-2
The flow volume loop in chronic obstructive pulmonary disease (emphysema). Flow in the vertical axis and volume is the horizontal axis. FEV1 is the forced expiratory volume in the first second of time. FVC is the forced vital capacity.
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tening of the expiratory curve, but it can be distinguished at times by a lack of reproducibility of successive curves. The report of the technician administering the test is perhaps the most helpful factor in distinguishing poor effort from disease, in many cases. Another frequent error in interpretation of expiratory curves is the presence of a knee (see Figure 2-3) high up on the curve, close to total lung capacity (TLC). This can be corrected by repeating the study while hyperextending the neck. The explanation is thought to be due to stiffening of the trachea and movement of the equal pressure point. In some younger patients, a “shoulder” can be seen further down the expiratory curve, where flows that were being maintained during the early part of the curve decrease suddenly and more steeply. In both these variants, however, a peak is visible. In healthy subjects, where these variants are usually seen, the peak flow rate is in the normal range, whereas with true tracheal disease, the rate is reduced. The inspiratory portion, in contradistinction to the expiratory portion of the FVL, is entirely effort dependent. Whereas the effort independent portion of the FVL is a function of elastic recoil of the lung as the driving force, the inspiratory portion is entirely driven by the force generated by the inspiratory muscles. Interpretation of this portion of the loop can be very difficult because many subjects without upper airway obstruction have difficulty in making a sustained forceful inhalation and technicians may not compel full efforts. For example, variable extrathoracic airway obstruction may be overdiagnosed, based solely on interpretation of the inspiratory portions of the loop.
Upper Airway Obstruction The presence of obstruction to airflow in the trachea can be evaluated by inspection of the FVL. Traditionally, this has been divided into fixed obstruction and mobile obstruction, and the latter subdivided into extrathoracic or intrathoracic obstruction. The term mobile is related to fluctuation of the diameter of the affected region under the influence of intraluminal or extraluminal pressure. In turn, these pressures are dependent on whether flow is occurring during inspiration or expiration.
FIGURE
2-3 Repetitive attempts at a flow volume loop, illustrating the importance of technique.
Physiology of the Trachea
Fixed Upper Airway Obstruction In fixed upper airway obstruction, both the inspiratory and expiratory loops demonstrate a plateau (see Figure 2-1B). Miller and Hyatt carried out experiments, in which forced exhalation and inspiration was performed through a series of progressively narrower tubes (Figure 2-4).1,2 These studies demonstrated that flattening of both inspiratory and expiratory curves occurred to an increasing degree with narrowing of the orifices. The features on the expiratory curve are first visible at about a 1-cm tracheal diameter, implying that there is a lack of sensitivity with mild narrowing of the trachea. However, once flow limitation begins, the reduction in flow rate is very rapid, with the peak flow rate falling from about 90% predicted at a 1-cm tracheal diameter to 25% at a 5-mm diameter. Both the length of the plateau and the degree of peak flow reduction are proportional to the degree of obstruction. Miller and Hyatt also evaluated the sensitivity of other standard pulmonary function tests in the diagnosis of tracheal obstruction (Figure 2-5).1,2 The peak expiratory flow rate was the most sensitive test, followed by the maximum voluntary ventilation. The forced expiratory volume in the first second (FEV1) does not show a recognizable fall outside of the normal range until at an approximately 6-mm tracheal diameter. The peak inspiratory flow rate is the most sensitive test for detecting inspiratory flow limitations. The ratio of maximum inspiratory flow (MIF) to maximum expiratory flow (MEF) at 50% vital capacity (MIF50/MEF50) remains about 1.0, since both parts of the FVL are altered to about the same degree.
FIGURE 2-4 Flow volume loops obtained from breathing through progressively smaller orifices. The solid line is the unobstructed loop. C = control. Reproduced with permission from Miller RD and Hyatt RE.2
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2-5 Inspiratory and expiratory flows obtained through progressively narrower external tubes. FEV1 = forced expiratory volume; FIV1 = forced inspiratory volume; MMF = maximum mid-expiratory flow; MVV = maximum voluntary ventilation; PEF = peak expiratory volume; PIF = peak inspiratory volume. Reproduced with permission from Miller RD and Hyatt RE.2
FIGURE
The most common cause of fixed stenosis is postintubation stenosis. The lesion is circumferential and usually related to mucosal and cartilage damage at the site of the cuff. Other causes are neoplasms, goiters, and stenosis of both main bronchi. Flow volume loops performed though a tracheostomy tube also show the same pattern. Gamsu and colleagues (Figure 2-6) derived a graphic representation of flow related to driving pressure through a series of fixed tubes of varying diameters.3 From this, and assuming a driving pressure of 100 cm H2O, the diameter of a stenotic segment may be estimated by measuring the MIF50 and MEF50. The method is reasonably accurate, within about a 1.5-mm tracheal diameter for rigid lesions, but it is less accurate when individuals have emphysema or marked malacic segments.
Variable Upper Airway Obstruction The term variable obstruction describes the situation where intraluminal and extraluminal pressures affect the anatomy of the lesion. This may occur because the involved area is malacic, or because the lesion, although firm, is not circumferential, and normal tracheal wall movement, usually the posterior membrane, alters the shape of the lumen. The alteration may also be postural, as in the case of a hemangioma.
Physiology of the Trachea
FIGURE 2-6 Graphic representation of airway diameter and flow (forced expiratory flow50 or forced inspiratory flow50) with airway pressure between 50 and 150 cm H2O, derived from progressively increasing external resistances in a normal subject. Reproduced with permission from Gamsu G et al.3
In variable extrathoracic obstruction (see Figures 2-1D, 2-7), the negative intraluminal pressure relative to the atmospheric pressure produced during inspiration causes narrowing of, for example, a malacic segment, resulting in limited inspiratory flow.4 During exhalation, a positive intraluminal pressure is generated, which results in maintenance or expansion of the diameter of the airway and a normal expiratory flow rate. Thus, a plateau is observed on the inspiratory loop alone, and the MEF50/MIF50 is greater than 1. Vocal cord paralysis, usually bilateral but also unilateral, is one of the most common causes of this pattern.5 The features are also seen in individuals with severe burns and vocal cord dysfunction. Variable intrathoracic obstructions (see Figures 2-1C, 2-8), such as tracheomalacia, demonstrate a reduction in the peak flow rate and a flattening of the expiratory loop, in response to the high positive intrapleural and, therefore, extraluminal pressures generated.4 The inspiratory loop is normal, responding to negative inspiratory pressures. The MEF50/MIF50 is low and the FEV1 less than the forced inspiratory volume in first second (FIV1). The major causes are malacic segments or noncircumferential tumors.
Upper Airway Obstruction and Chronic Obstructive Pulmonary Disease Tracheal abnormalities frequently develop in the presence of chronic obstructive pulmonary disease (COPD). This may be due to injuries, such as cicatrization at the site of an endotracheal cuff producing fixed stenosis, tracheomalacia at the site of an endotracheal cuff leading to a variable intrathoracic stenosis, or variable extrathoracic stenosis due to vocal cord dysfunction or malacia at the site of a tracheostomy. The differentiation between the effect of COPD and tracheal injury is important because (a) recognition can lead to repair and (b) it is important to understand the relative contribution of the stenosis and COPD to symptoms such as dyspnea. For example, the presence of a minor degree of radiographically and bronchoscopically recognizable stenosis may have little significance in the presence of very severe COPD, where the flow limitation is mainly in the distal airways, hence the stenosis does not provide any further impediment to flow nor to expectoration of secretions.
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FIGURE 2-7
Inspiration and expiration influencing variable extrathoracic obstruction. Patm is the atmospheric pressure; Ptr is the intratracheal pressure. Reproduced with permission from Kryger M et al.4
FIGURE 2-8
Inspiration and expiration influencing variable intrathoracic obstruction. Ppl is the intrapleural pressure; Ptr is the intratracheal pressure. Reproduced with permission from Kryger M et al.4
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Fixed tracheal stenosis, variable intrathoracic stenosis, and COPD all reduce the peak flow rate. However, the tracheal lesions produce a plateau, whereas a sharp peak is still seen in COPD, together with concave expiratory flow patterns. The length of the plateau and the reduction of peak flow by the plateau, as described earlier, are approximately proportional to the severity of the stenosis. As exhalation proceeds, the site of flow limitation moves peripherally until the collapsing small airways inhibit flow. At that point, the concave pattern of COPD will become the visible feature (Figure 2-9). Thus, the ratio of plateau to concavity gives an approximation of the relative significance of the lesions. If no plateau is visible, the stenosis is not contributing to flow limitation, nor is it likely to contribute to dyspnea. However, the reverse is not necessarily true; that is, where stenosis is contributing to flow limitation, release of stenosis may not relieve dyspnea much, since other factors such as hyperinflation related to the residual COPD or the presence of cor pulmonale may be very important in the mechanism of dyspnea. However, improvement in the ability to cough up secretions may have a significant effect. Thus, COPD may conceal stenosis, and stenosis may cause one to underestimate the severity of COPD. In addition, the presence of signs or symptoms also depends on the relationship between the flow required for a specific activity and the degree of obstruction. Geffin and colleagues demonstrated that, at rest, stridor was only heard when the airway was reduced to a 5 mm stenosis.6 However, with exercise, a less severe stenosis may also cause stridor.7 Another useful technique is the superimposition of the tidal volume loop on the FVL, with tidal volume expressed both at rest and during hyperventilation. The point of contact of the tidal volume with inspiratory or expiratory plateaus or with the concavity of COPD provides very useful information about the limiting factors and the contribution of the plateau to limiting flow and to the symptoms. Accurate positioning of the tidal volume on the axis can however be difficult. Helium-oxygen inhalation, which reduces the density of the gas, has also been used to differentiate COPD from upper airway obstruction (UAO).8 Since flow in the upper airway is density dependent, helium-oxygen inhalation can increase expiratory flow in the UAO, but not in COPD when laminar flow is much less dependent on density.9 Nitrogen washout using 100% oxygen is normal in UAO, but abnormal in COPD10,11; however, this is mainly useful when pure forms of these processes are present, whereupon an FVL should suffice.
April 11
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April 18
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2-9 Flow volume loops of an individual with chronic obstructive pulmonary disease (COPD), taken prior to tracheal stenosis and a bronchodilator (A), with bronchodilator, where flow increases sufficiently to demonstrate a plateau due to concomitant tracheal stenosis (B), and progression of the tracheal stenosis, concealing much of the COPD (C). V= flow; VC = vital capacity. FIGURE
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Miller and Hyatt presented data suggesting that the ratio of the mid-expiratory flow rate (MEF50) to the mid-inspiratory flow rate (MIF50) would enhance the visual evaluation of the FVL.2 A normal ratio would be from 0.9 to 1.0. A fixed obstruction, with both loops limited, would remain at about 0.9, whereas a variable extrathoracic obstruction would be greater than 1.0, and a variable intrathoracic obstruction would be 0.2 or less. These values for variable obstruction relate mainly to severe disease. The dorsal membrane of the trachea is invagin*ted to some degree in healthy individuals when exposed to high intraluminal pressures, such as may occur with a cough or during exercise. In some individuals with severe obstructive lung disease, this invagin*tion as visualized at bronchoscopy may be so severe as to almost completely obliterate the lumen. The effect is to markedly limit the ability of individuals to clear secretions via coughing. This may be particularly important in patients with bronchitis extending to their distal airways. Herzog and colleagues investigated a group of these patients using a technique of intrabronchial pressure measurements, slowly withdrawing a catheter from distal to central airways and measuring simultaneous alveolar pressure and flow rate in a plethysmograph.12 They calculated the flow resistance over bronchial segments and found patients with collapse of the central airways, as well as those with collapse of both peripheral and central airways, when exposed to high intrapleural pressure. These findings were correlated with bronchoscopic inspection, under local anesthesia, of the posterior membrane being invagin*ted by high intrapleural pressure induced by coughing and hyperventilation. Herzog and colleagues described a typical appearance of a spirometric tracing, in which there is a sudden fall in the flow rate, known as the “check valve” phenomenon. In an individual with severe COPD, this can also be seen in an FVL as a sudden fall in expiratory flow, followed by a slow further decline in flow (Figure 2-10). The posterior membrane was stabilized by grafting fascia or plastic material. Herzog and colleagues showed a marked increase in FEV1 in some cases, although not all. There was a general improvement in the partial pressure of oxygen (PO2) and a fall in the partial pressure of carbon dioxide (PCO2) where this was initially elevated. The authors were careful to point out that these results did not mean that the collapse of airways in COPD was solely in the central airways; the small airways were still the major site of collapse. However, the ability to clear secretions and limit the number of episodes of bronchitis and reactive airway constriction was probably the most important factor in the improvement. They noted that this process was of most assistance in individuals with a clinical syndrome of chronic bronchitis and severe attacks of coughing even up to the point of cough syncope. This is a concept that probably needs to be re-addressed. Certainly, the results in the small number of cases described appear to at least equal the effect of volume reduction surgery on the FEV1. The effect of exercise in patients with tracheal stenosis has been sparsely studied. This is a difficult task because many patients with tracheal stenosis also have parenchymal disease, due to coexistent problems such as chronic obstructive lung disease, asthma, or bronchiectasis, which influences the results. Seven patients without diffuse lung disease were studied using mild exercise,7 since patients with tracheal stenosis are limited in their ability to exercise by dyspnea. In all patients, the PO2 decreased with exercise, with the mean being 11 mm Hg. In general, the magnitude of PO2 decrease correlated with the degree of obstruction. PCO2 only increased an average of 2 mm Hg. In the three subjects who had surgical correction, the PO2 rose by a mean of 9 mm Hg and the PCO2 fell slightly with exercise. The vital capacity was not changed by corrective surgery, but the FEV1, maximum breathing capacity, and peak expiratory flow rate all increased markedly.
Physiology of the Trachea
FIGURE 2-10 Flow volume loop showing a sudden dramatic decline in expiratory flow followed by a long plateau, normal inspiratory loop because of the effect of a negative inspiratory intrapleural pressure on the floppy airways. Flow is the vertical axis and volume is the horizontal axis. FEV, is the forced expiratory volume at first second. FVC is the forced vital capacity.
References 1. 2. 3. 4. 5. 6.
Miller RD, Hyatt RE. Evaluation of obstructing lesions of the larynx and trachea by flow-volume loops. Am Rev Respir Dis 1973;108:475–81. Miller RD, Hyatt RE. Obstructing lesions of the larynx and trachea: clinical and physiological characteristics. Mayo Clinic Proc 1969;44:145–61. Gamsu G, Borson DB, Webb WR, et al. Structure and function in tracheal stenosis. Am Rev Resp Dis 1980;121:519–31. Kryger M, Bode F, Antic R, et al. Diagnosis of obstruction of the upper and central airways. Am J Med 1976;61:85–93. Cormier Y, Kashima H, Summer W, et al. Airflow in unilateral vocal cord paralysis before and after Teflon injection. Thorax 1978;33:57–61. Geffin B, Grillo HC, Cooper JD, Pontoppidan H. Stenosis following tracheostomy for respiratory care. JAMA 1971;216:1–8.
7.
8.
9.
10. 11.
12.
Al-Bazzaz F, Grillo H, Kazemi H. Response to exercise in upper airway obstruction. Am Rev Respir Dis 1975;111:631–40. Lavelle TF Jr, Rotman HH, Weg JG. Isoflow-volume curves in the diagnosis of upper airway obstruction. Am Rev Respir Dis 1978;117:845–52. Gold M, Marks A, Bocles JS. Effects of reduction in air density on dynamic function in obstructive airways disease. Am Rev Respir Dis 1964;90:316–20. Sackner MA. Physiologic features of upper airway obstruction. Chest 1972;62:414–7. Simonsson BG, Malmberg R. Differentiation between localized and generalized airway obstruction. Thorax 1964;19:416–9. Herzog H, Keller R, Allogower M. Special methods of diagnosing and treating obstructive diseases of the central airway. Chest 1971;60:49–67.
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CHAPTER THREE
Pathology of Tracheal Tumors 3A EPITHELIAL TUMORS OF THE TRACHEA Javad Beheshti, MD Eugene J. Mark, MD Fiona Graeme-Cook, MD
Squamous Epithelial Tumors Adenocarcinoma Large Cell Undifferentiated Carcinoma Neuroendocrine Tumors
Salivary Gland-Type Tumors Malignant Melanoma Metastatic Tumors to the Trachea
Epithelial proliferations of the trachea, like the bronchus and lung and many other organs, comprise the common tumors obstructing the trachea. As with the bronchus and lung, the majority of epithelial neoplasms in the trachea are malignant, although the frequency of carcinomas in the trachea is much less. This lowered frequency may be due to the limited surface area of the epithelium, greater mucociliary stream, and more laminar airflow compared to the bronchial tree. Normally, the trachea is lined by columnar ciliated respiratory epithelium, which has both mucinous and nonmucinous cells. Many neoplasms of the trachea, both benign and malignant, are squamous rather than mucinous or glandular, reflecting the conversion of respiratory epithelium to squamous epithelium under noxious stimuli, particularly cigarette smoke. The squamous proliferations resemble those in the lung in many regards. The glandular neoplasms can resemble either tumors of the lung or salivary gland, the latter because the mucus glands in the trachea are similar to mucinous glands and ducts in the oropharynx and salivary glands. Because tumors of the trachea obstruct this single vital structure, detection occurs when the tumor is at a smaller size compared to the average for a pulmonary tumor, and metastasis even to regional lymph nodes is not common. Nevertheless, difficulties in resecting tracheal lesions adversely affect the prognosis in some patients, where recurrent tumor develops after incomplete resection or, in the rare case, where local metastasis has already occurred.
Squamous Epithelial Tumors Squamous Papilloma and Papillomatosis Squamous papillomas of the trachea are rare benign tumors composed of stratified squamous epithelium with acanthosis and papillomatosis, supported by a fibrovascular core. They are either multiple and recurrent (papillomatosis) or solitary exophytic growths into the tracheal lumen.
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Biology. Squamous papillomatosis occurs as multiple and recurrent squamous cell papillomas of the trachea, often associated with upper (mostly laryngeal) and/or lower (bronchial) involvement. Children and adolescents are most commonly affected, hence the term “juvenile papillomatosis”; occasional adults with disease have been reported.1 The patient may present with stridor, wheezing, dyspnea, chest pain, or hemoptysis. The lung parenchyma is affected in about 1% of cases, most of them complicated by necrosis, cavitation, or pneumonia. The disease sometimes regresses in adulthood, but the course is usually protracted in extensive papillomatosis with complications of obstruction in the trachea, larynx, or bronchi, with atelectasis, bronchiectasis, and pneumonia. Surgical resection, laser fulguration, and interferon therapy are usually attempted to secure patent airways in these patients. Human papilloma virus (HPV) is a known cause of these tumors, and different types are detected. HPV types 6 and 11 are commonly found in benign lesions, whereas types 16 and 18 are mostly associated with malignant transformation.2 Other types including 31, 33, and 35 are also occasionally found in malignantly transformed cases.2 Solitary papillomas often occur in adults, but children may also have solitary tumors. Squamous cell carcinomas may develop in about one-third of solitary squamous papillomas without other risk factors.3 In contrast, malignant transformation is less common in multiple papillomas (papillomatosis), occurs on average about 15 years after initial diagnosis, and is often associated with risk factors such as radiation, cytotoxic drug therapy, and smoking.4 Pathology. Grossly, the papillomas appear as cauliflower-like excrescences, protruding into the tracheal lumen (Figures 3-1, 3-2 [Color Plate 1]). Microscopically, papillomas are composed of loose fibrovascular cores covered by hyperplastic stratified squamous epithelium with papillomatosis. Keratinization may be present sometimes with small parakeratotic foci. Koilocytosis (perinuclear halo and nuclear wrinkling) is seen in all papillomatosis cases and in solitary papillomas associated with HPV. The epithelium is usually cytologically bland; mitoses, atypicality, or dysplastic cells are infrequent, but are occasionally seen in solitary papillomas (Figure 3-3 [Color Plate 1]). A case of a cytologically benign papilloma was reported in a 27-year-old man, where the tumor invaded the tracheal wall and adjacent soft tissues, without nodal or distant metastasis in 4 years of followup. The term “invasive tracheal papillomatosis” was suggested in this case.5 Papillomas must be differentiated grossly and microscopically from papillary squamous cell carcinomas, which they resemble because of papillary configuration and layers of neoplastic squamous cells, but lack similar atypical cellular and architectural features. Squamous cell carcinomas may also arise in papillomas, showing focal cellular pleomorphism, loss of maturation, dyskeratosis, and increased hyperkeratosis. It then extends through the epithelium and ultimately invades the underlying connective tissue. It may go through the tracheal wall into adjacent soft tissues and lymphatics. These can be well or moderately differentiated, with or without keratinization. Small endoscopic biopsies can result in improper interpretation, since malignant transformation can be well differentiated or focal. Conversely, some focal atypia have been observed in benign papillomas.6 Extension of squamous epithelium to bronchial glands should not be interpreted as invasion.
Squamous Cell Carcinoma In many published series, squamous cell carcinoma is the most common primary tracheal tumor, whereas in others, it equals in frequency or comes after adenoid cystic carcinoma as the second-most common neoplasm in adults.7–9
Pathology of Tracheal Tumors
Biology. Although one of the two most common tracheal malignancies, squamous cell carcinoma is far less common than its laryngeal or bronchogenic counterparts, which are about 75 and 180 times more frequent, respectively.10 Age distribution is between 20 and 80 years with peak incidence in the sixth and seventh decades, similar to bronchogenic squamous cell carcinoma. An infant with tracheal squamous cell carcinoma has been reported.8 Men are significantly more affected than women, with a ratio of 2:1 to 4:1 in large series.8,9 Smoking history is present in most patients, accounting for almost all cases in some series, suggesting a strong association as in bronchogenic squamous cell carcinoma.7 The lower incidence of tracheal squamous cell carcinomas compared with those of the bronchus is attributed by some to laminar airflow in the trachea (because of large diameter) and effective mucociliary clearance (because of evenness and absence of bifurcation). These may prevent accumulation of carcinogens in the mucosa which can promote a malignant transformation sequence.11 Six patients have been reported with tracheal squamous cell carcinoma arisen from tracheostomy scars, probably the result of carcinogenesis in active repair, similar to scar carcinoma elsewhere.12 One case was reported in a plumber exposed to asbestos.13 Most cases of primary squamous cell carcinoma occur as solitary lesions, but synchronous and metachronous tumors, mostly with bronchogenic, laryngeal, or esophageal squamous cell carcinomas, have been reported.14,15 Pathology. Grossly, squamous cell carcinomas usually arise from the posterior tracheal wall as polypoid growths, most commonly in the lower third, followed by the upper third (Figures 3-4, 3-5 [Color Plate 1]).8 Surface ulceration is often present. Microscopically, the tumor is composed of nests and sheets of squamous cells with various degrees of differentiation (Figures 3-6, 3-7, 3-8 [Color Plate 1]). Squamous differentiation, as in the lung, includes intercellullar bridges and keratin pearl formation. Two cases of basaloid variants have been reported, wherein like the cutaneous basal cell carcinoma, the cells are smaller, with scant cytoplasms and high nuclear to cytoplasmic ratios.16 Combined squamous cell and small cell carcinomas have also been reported.17 The differential diagnosis includes papilloma, squamous cell carcinoma arising in papilloma, mucoepidermoid carcinoma, and necrotizing sialometaplasia. It must also be distinguished from squamous cell carcinomas invading from adjacent structures such as the esophagus, lung, thymus, and even thyroid gland. These primaries must always be considered when squamous cell carcinoma is diagnosed by endoscopic biopsy.
Adenocarcinoma Although the most common lung malignancy, adenocarcinomas are usually located in the lung periphery, away from the central airways; they are infrequent in central bronchi and much rarer in the trachea. Biology. Adenocarcinoma has been reported in several recent series of primary tracheal tumors as a rare entity.9,18 However, in older literature, its occurrence was much higher, and exceeded squamous cell carcinoma as the most common tracheal malignancy in some series.19 It is not clear whether some cases of other primary tumors, such as mucoepidermoid carcinoma or adenoid cystic carcinoma, adenocarcinomas invading from adjacent structures, and metastatic adenocarcinomas, had been misclassified as primary adenocarcinomas in those series, and hence partly responsible for the high incidence of this tumor in older publications. Adenocarcinoma affects both men and women, and occurs mostly in the fifth to eighth decades of life. Presentation is often with obstructive symptoms. One patient presented with a thyroid mass, later found to be invasion from tracheal adenocarcinoma.20
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Pathology. Grossly, adenocarcinomas are bulky tumors that may bulge into the lumen, but they also invade through the tracheal wall into adjacent structures. Microscopically, most of the reported cases have been of the mucin-producing category. They form glands, with the epithelial cells containing large vesicular nuclei and prominent nucleoli (Figures 3-9, 3-10 [Color Plate 2]). Differential diagnosis includes adenoid cystic carcinoma, mucoepidermoid carcinoma, and secondary involvement of trachea from other primaries. The latter includes direct invasion from thyroid gland carcinomas and adenocarcinomas of proximal bronchi extending up to the trachea. Metastatic adenocarcinomas must be distinguished from tracheal primaries, as cases have been reported from colorectal adenocarcinomas and endometrial adenoacanthomas.21,22 A case of adenocarcinoma ex pleomorphic adenoma (arising in pleomorphic adenoma) of the trachea has been reported.23 Also reported are cases of thyroid carcinoma arising from ectopic thyroid tissue in the trachea.24 Various degrees of histologic differentiation in tracheal adenocarcinomas remain to be clarified, since this description is not given in most of the published series. Tracheal adenocarcinomas seem to follow an invasive and rapidly fatal course; however, valid survival analyses have not been performed so far.
Large Cell Undifferentiated Carcinoma Also called “large cell carcinoma” or “anaplastic carcinoma,” this malignant epithelial tumor consists of sheets of cells with large nuclei, prominent nucleoli, abundant cytoplasm, and usually well-defined cell borders, without the characteristic features of squamous cells, small cells, or adenocarcinomas.25 Biology. Large cell carcinoma of the lung almost always occurs in smokers with a median age of 60 years.26 Its incidence in the trachea ranges from none in some series to as high as 21% in others.11,27 Pathology. Grossly, these tumors are usually large tan or white necrotic masses, sometimes with hemorrhage. Microscopically, sheets of large polygonal cells with large vesicular nuclei and prominent nucleoli are observed. Cytoplasm is abundant and the cell membrane is usually prominent. Squamoid features may be seen, but keratin pearls and intercellular bridges are lacking. Gland formation and mucin production, which may be identified by mucin stains, are not present. Therefore, the diagnosis is made by exclusion of squamous cell carcinoma and adenocarcinoma. Small endoscopic samples may lack foci of differentiation that are evident later in a resected specimen. When neuroendocrine differentiation is suggested by immunohistochemical or electron microscopic examinations, the tumor corresponds to “large cell carcinoma with neuroendocrine differentiation” as occurs in the bronchi. It is distinguished from “large cell neuroendocrine carcinoma,” which shows an organoid pattern, rosetting, and peripheral palisades. Differentiation from small cell carcinoma is evidenced by large vesicular nuclei, prominent nucleoli, abundant cytoplasm, and absence of nuclear molding. Smearing (crushing) artifact is more usual for small cell carcinoma. Histological variants include giant cell carcinoma, spindle cell carcinoma, pleomorphic (giant cell/spindle cell) carcinoma, clear cell carcinoma, and lymphoepithelioma-like carcinoma. Only giant cell carcinoma and lymphoepithelioma-like carcinoma have been reported in the trachea.28,29 Giant cell carcinoma has large cells (ie, two or three times the size of classic large cell carcinomas) and huge, bizarre, and pleomorphic giant cells. Giant cells may be seen focally in many lung carcinomas and may also occur after radiation therapy of tumors, and a prominent component of giant cell change (ie, above 10% of tumor cells) is necessary to make the diagnosis.25,28 It is not certain whether the reported giant cell carcinomas in the trachea meet the given criteria. The only reported case of lymphoepitheliomalike carcinoma had similarity to the so-called nasopharyngeal lymphoepithelioma.29
Pathology of Tracheal Tumors
Mixed small cell/large cell carcinoma has been seen in the lung, and a case of mixed small cell/squamous cell/giant cell carcinoma in the trachea has been reported.30 Anaplastic or undifferentiated carcinoma invading the trachea from adjacent structures must always be considered in the differential diagnosis of primary tracheal large cell carcinoma. In a series of anaplastic thyroid carcinoma, tracheal invasion was reported in some cases.31
Neuroendocrine Tumors This group of malignant neoplasms comprises a spectrum from low-grade typical carcinoid to high-grade neuroendocrine tumors. These tumors are much less frequent in tracheae than in bronchi, probably due to a scarcity of Kulchitsky cells (neuroendocrine cells of origin) in the trachea. Bronchopulmonary neuroendocrine tumors are classified into low-grade typical carcinoid, intermediate-grade atypical carcinoid, and high-grade carcinoid categories of large cell neuroendocrine carcinomas and small cell carcinomas. Because of their rarity in the trachea and lack of large studies, some of the data given here are based on bronchial counterparts, which probably do not differ much from tracheal ones.
Typical and Atypical Carcinoid Tumors Carcinoid tumors are low-grade malignant neoplasms of neuroendocrine cells. They are divided into typical and atypical subtypes, with the latter possessing more malignant histologic and clinical features.32 Biology. Carcinoid tumors are reported from childhood to old-age.33 The median age for patients with bronchial carcinoid tumor is 55 years, and it is the most common bronchial tumor of childhood.34 Males and females are equally affected. Pathology. Grossly, carcinoid tumors usually have a main polypoid intraluminal component, with a smooth and tan-yellow to pink cut surface (Figure 3-11 [Color Plate 2]). Occasionally, they can be virtually confined to the polyp and have only minor growth in the lamina propria of the tracheal wall. Atypical carcinoids may be more infiltrative through the wall, sometimes with areas of necrosis or hemorrhage. Local lymph node metastasis is frequently seen in atypical carcinoid tumors. Microscopically, both typical and atypical carcinoids show the so-called “neuroendocrine look,” which is an organoid pattern of uniform epithelial cells with a finely granular chromatin pattern, inconspicuous nucleoli, and moderate eosinophilic cytoplasm (Figures 3-12, 3-13, 3-14 [Color Plate 2]). Atypical carcinoids have coarser chromatin with more prominent nucleoli. Other histologic patterns are those that are trabecular, glandular, paraganglioma-like, rosette-like, and palisaded. Usually, more than one pattern is seen in a given tumor. Uncommon patterns seen in tracheal carcinoids are those that are oncocytic and melanin-producing.35 It is well known that atypical carcinoids are cytologically more atypical with a higher mitotic rate and necrosis, but the criteria of separation from typical carcinoids have been challenging over the years. The most recent criteria were proposed by Travis and colleagues for bronchopulmonary carcinoids, and seem to be reproducible and well correlated with clinical behavior and survival.36 Based on these criteria, atypical carcinoids are characterized by a mitotic rate of 2 to 10 per 10 high-power fields or foci of coagulative necrosis; that is, either punctate or large and infarct-like. Typical carcinoids are often well delimited or have a broad front of invasion. Atypical carcinoids are more likely to have tongues of invasion into the tracheal wall. Vascular and lymphatic invasions are common in atypical carcinoids. Differential diagnosis includes small cell carcinoma and undifferentiated large cell carcinoma, which can be very difficult on interpretation of small or crushed endoscopic biopsies. Depending on
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the various patterns present in carcinoid tumors, other tumors may be considered in the differential diagnosis, but it is not clear how often uncommon patterns may be seen in tracheal carcinoids.37 An oncocytic pattern must be separated from other forms of oncocytic tumors. A glandular pattern may be difficult to differentiate from adenoid cystic carcinoma and mucoepidermoid carcinoma. Important is the absence of hyaline cores and mucin, respectively. Immunohistochemical studies for neuroendocrine markers are often necessary on a small biopsy. Melanocytic carcinoid tumors must be separated from melanomas. Metastatic carcinoma of the trachea is another group in the differential diagnosis. The 5- and 10-year survival rates for typical bronchial carcinoid tumors are both at 87%, and for atypical carcinoid tumors, are at 56% and 35%, respectively.36 The survival for atypical carcinoid tumor of the trachea is probably less than its bronchial counterpart, because local lymph node metastasis for this midline organ can be bilateral and difficult to encompass surgically at the outset.
Large Cell Neuroendocrine Carcinoma Large cell neuroendocrine carcinoma (LCNEC) is a high-grade neuroendocrine tumor, which may be mistaken pathologically for a carcinoid or atypical carcinoid tumor, but has a worse prognosis. This tumor shows a light microscopic, immunohistochemical, and ultrastructural neuroendocrine differentiation. Biology. There is only one published study on these tumors in the trachea.38 This could be due to its rarity, but it may also be due to misclassification as an atypical carcinoid or large cell carcinoma. The data and descriptions that follow are mainly from pulmonary LCNECs. The patient ages range from 35 to 75 years, and the tumor occurs mostly in cigarette smokers.39 Pathology. Grossly, the cut surface is usually yellow, tan, or white, with necrosis. Hemorrhagic foci may be present. Microscopically, the tumor shows neuroendocrine features with organoid, palisading, trabecular, or rosette-like growth patterns. It usually has nests of tumor cells with central necrosis. The cells are large with vesicular nuclei and prominent nucleoli. The mitotic rate is more than 10 per 10 high-power fields. Endocrine differentiation may be shown on immunohistochemical or ultrastructural studies. The main differential diagnosis is of large cell carcinoma with neuroendocrine features. This distinction is made by light microscopic evidence of a “neuroendocrine look,” such as an organoid pattern, peripheral palisades, or rosetting in LCNEC. Immunohistochemistry and electron microscopy are not helpful since both have neuroendocrine granules. Atypical carcinoid is separated based on mitotic activity (less than 5 per 10 high-power fields). Small cell carcinoma is sometimes difficult to differentiate, particularly when the cells are larger in small cell carcinomas or when the biopsy is small and crushed. Overall, smaller cells in small cell carcinomas, as well as nuclear molding, a fine chromatin pattern, absence of inconspicuous nucleoli, a smearing effect, and basophilic staining of vessels (Azzopardi effect) are helpful distinguishing features. The prognosis is worse than for atypical carcinoids, and approaches that for small cell carcinoma.38 The response to chemotherapy seems to be better than that of nonsmall cell carcinomas.
Small Cell Carcinoma Small cell carcinoma is the other high-grade neuroendocrine carcinoma of the tracheobronchial tree. Biology. Relatively common among bronchial malignancies, small cell carcinoma has been regarded as a very rare tumor in the trachea. In most studies, it is either not present or is reported as one of the least
Pathology of Tracheal Tumors
common tracheal malignancies. However, in two of the largest series with 321 and 43 cases, it comprised 6% and 7% of tracheal tumors, respectively.18,40 Hormonal activity was reported in a case with both oncogenic hypophosphatemia and ectopic corticotropin production causing Cushing’s syndrome.41 Pathology. In bronchi, small cell carcinoma typically infiltrates the wall, with irregular thickening of mucosa, and it less commonly produces a tapered narrowing or intraluminal mass. When there is extensive paratracheal small cell carcinoma, it may be difficult to know whether the tumor arose in the hilum of the lung, esophagus, thymus, or trachea. Microscopically, the tumor consists of irregular islands of small cells, about two or three times the size of lymphocytes, with a high nuclear to cytoplasmic ratio. Nuclei are round to oval with hyperchromatic finely-granular chromatin, nucleoli are absent or inconspicuous, and cytoplasm is scant (Figures 3-15, 3-16 [Color Plate 2]). The cells usually lie close to each other, sometimes with nuclear molding. Extensive or punctate necrosis is common and mitotic figures are frequent. Less common morphologic features include slightly larger polygonal cells with more cytoplasm, formation of tubules and rosettes, and peripheral palisading, all of which are seen in bronchial small cell carcinomas. The neuroendocrine nature of the tumor cells is occasionally demonstrable by immunohistochemical studies with neuroendocrine markers, such as chromogranin, synaptophysin, neuron-specific enolase (NSE), and neuron cell adhesion molecule (N-CAM), or as dense core granules on electron microscopy. In practice, however, these studies may be difficult to interpret in a small crushed biopsy. A case of mixed squamous and small cell carcinoma and another of mixed squamous cell, small cell, and giant cell carcinoma have been reported in the trachea.17,30 Differential diagnosis includes other neuroendocrine tumors (carcinoid, atypical carcinoid, large cell neuroendocrine carcinoma), nonsmall cell carcinoma (adenocarcinoma, squamous cell carcinoma, large cell carcinoma, whose cells are sometimes relatively small), small round cell tumors (such as lymphoma, embryonal rhabdomyosarcoma), and adenoid cystic carcinoma. The diagnosis may be particularly difficult in small endoscopic biopsies due to crushing artifact of cells, which can also occur in lymphocytic infiltration and neuroendocrine and other tumors. The exclusion of malignant lymphoma by cytology or immunohistochemical markers is particularly important because of the different therapy and prognosis for tracheal lymphoma. Similar to bronchial small cell carcinoma, this tumor has the worst prognosis among the tracheal malignancies.
Salivary Gland-Type Tumors The minor salivary glands existing in the mucous membranes of the head and neck extend downward as the submucosal glands in the trachea. Therefore, they are subject to most of the so-called “salivary glandtype tumors” that involve the salivary glands of the head and neck, with only few exceptions. However, the frequency of such tumors is much lower in the trachea than in the head and neck region.
Pleomorphic Adenoma (Mixed Tumor) Pleomorphic adenoma is primarily a benign tumor of the major salivary glands. Rarely, it can arise from seromucous glands of the tracheobronchial tree. Biology. Approximately 30 cases of tracheal pleomorphic adenoma have been reported in the literature. The male to female ratio is about 2:1 with an age range of 15 to 80 years. Over one-half of patients were in the fifth and sixth decades of life.
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Pathology. The upper third of the trachea is the most common site of origin, followed by the middle and lower thirds.42 The tumor typically grows as a polypoid intraluminal mass, causing various degrees of obstruction (Figure 3-17 [Color Plate 3]). Microscopically, pleomorphic adenoma is relatively well circ*mscribed, but without a capsule. It consists of a mixture of epithelial cells, myoepithelial cells, and stroma (Figures 3-18, 3-19 [Color Plate 3]). The epithelial component forms sheets, ducts, trabeculae, and small nests of cells with vesicular nuclei and small to moderate amounts of cytoplasm. Foci of squamous differentiation may be seen. These areas merge with alternating areas of spindled and stellate cells in myxoid, hyaline, or chondromyxoid stroma. The terms “pleomorphic” and “mixed” both describe the contribution of epithelial and mesenchymal elements, but one or the other may predominate, almost to the exclusion of the other. Differential diagnoses include other salivary gland-type tumors like adenoid cystic carcinoma and mucoepidermoid carcinoma. Polymorphous low-grade adenocarcinoma, which sometimes is a difficult tumor to distinguish from pleomorphic adenoma in salivary glands, has been reported in the bronchi but not in the trachea. The prognosis is excellent, with only one recurrence reported. One patient died of tracheal obstruction. A case of carcinoma arising in pleomorphic adenoma (carcinoma ex pleomorphic adenoma) has also been reported, and we have seen a case where the majority of the tumor was pleomorphic adenoma, but the invasive tumor in the adventitia was carcinoma.23
Mucous Gland Adenoma Mucous gland adenoma is a benign tumor of salivary gland-type, which can rarely arise in seromucous glands of the tracheobronchial tree. A small number of cases has been reported in bronchi and two cases in the trachea.43,44 This tumor is characterized by cystically dilated mucous-filled glands, hence the synonymous term “mucous gland cystadenoma.” The cystic glands are lined by monomorphic cuboidal to columnar cells. Papillary proliferation can be present in the luminal surface of the tumor in both bronchial and tracheal mucous gland adenomas. The major differential diagnosis is mucoepidermoid carcinoma, which in low-grade cases could be predominantly glandular with a banal cytology. The presence of intermediate cells in mucoepidermoid carcinoma helps in this distinction.
Adenoid Cystic Carcinoma Called “cylindroma” in the past because of the production of cylindrical structures by the tumor cells, adenoid cystic carcinoma is one of the two most common tracheal malignancies, and together with squamous cell carcinoma, accounts for more than two-thirds of all tracheal tumors.9,18,45 Although it is less common than its head and neck counterpart, its occurrence is higher than bronchial adenoid cystic carcinomas. Biology. The age distribution for patients with this tumor is generally between 15 and 80 years, with the peak incidence occurring in the fifth decade, and the mean age being 10 years less than patients with squamous cell carcinoma.9,45,46 The sex distribution is almost equal.9,45,46 No association with smoking has been reported. The slow-growing nature of this malignancy causes late symptoms, which adds to the general diagnostic delay of tracheal tumors due to anatomic characteristics of the trachea (mainly its large luminal diameter) and often unhelpful chest x-rays. Pathology. Grossly, the tumor grows as a polypoid intraluminal mass or infiltrates the wall to cause thickening which can be vertical, horizontal, or circumferential, with eventual narrowing of the lumen (Figures 3-20 through 3-23 [Color Plate 3]).47
Pathology of Tracheal Tumors
Histologically, the tumor consists of small basaloid cells with a relatively high nuclear to cytoplasmic ratio and scant cytoplasm (Figure 3-24 [Color Plate 3]). The nuclei are round to oval, and dark and monotonous. Three histologic subtypes are identified: cribriform, tubular, and solid (Figures 3-25, 3-26, 3-27 [Color Plate 4]). The most common is the cribriform type, in which the neoplastic cells are arranged in nests and sheets fenestrated by round to oval spaces. These spaces contain eosinophilic periodic acid-Schiff (PAS)-positive basem*nt membrane-like material. The tubular subtype has single-lumen tubular units with smaller nests than the cribriform subtype. In the solid pattern, the cells are packed together to form nests and sheets with few lumina. Commonly, more than one pattern is present in a given tumor. The overlying epithelium is usually intact, with the tumor infiltrating submucosal tissue, going through the wall to adjacent structures, particularly from the posterior wall, where there is no cartilage. The neoplastic cells have a marked tendency for perineural invasion (Figure 3-28 [Color Plate 4]). Because of this infiltrating nature, microscopic examination often shows tissue involvement beyond grossly visible or palpable tumors. Histological grading is based on the presence and percentage of solid pattern, which inversely affects prognosis.48 Grade 1 is a tumor composed of tubular and cribriform subtypes, without any solid areas. Grade 2 consists of tubular and cribriform subtypes, with less than 20% solid subtype. In Grade 3, the solid subtype comprises more than 20% of the area of the section. This grading system has been shown to correlate with tumor behavior and patient prognosis.48,49 Differential diagnosis includes other carcinomas, mostly adenocarcinomas. Solid areas with few cystic spaces may be confused with adenocarcinomas, particularly since the nuclei can be larger and vesicular in poorly differentiated adenoid cystic carcinomas. In cases in which the morphology is close to adenocarcinoma with large sheets and few cystic or glandular spaces, the behavior is likely to be that of an adenocarcinoma. Small cell carcinoma must also be differentiated from the solid subtype. The more even chromatin pattern, nuclear crowding, and molding in small cell carcinoma are of help in this distinction. Frozen section examination of proximal, distal, and circumferential resection margins are essential at the time of definitive resection because of the surreptitious manner in which this tumor spreads. Two potential oversights are noteworthy. First, nests of tumor the size of lobules of normal tracheal mucous glands may partially replace the normal lobules without altering the overall mucosal architecture, thus escaping detection on casual examination. Second, perineural infiltration by individual cells may be the only tumor present in peritracheal adventitia. The paratracheal nerves deserve identification. Adenoid cystic carcinomas tend to be locally recurrent, particularly with inadequate safe margins, but nodal paratracheal metastases develop late in the course. Distant hematogenous metastases are infrequent in our experience, and fatal complications are more commonly due to recurrent tumor. When they occur, they are usually in the lung, liver, or bone. The prognosis is much better than squamous cell carcinoma, with 5- and 10-year survivals being reported to be about 75% and 50%, respectively.
Mucoepidermoid Carcinoma Mucoepidermoid carcinoma is a malignant salivary gland-type neoplasm arising from submucosal glands of the trachea. Until its histological description and definition as a separate entity in the tracheobronchial tree by Smetana and colleagues in 1952, and by Liebow 1975, and even years thereafter, it was classified as a “bronchial adenoma” together with carcinoid tumor and adenoid cystic carcinoma.50,51 Biology. Mucoepidermoid carcinoma is a rare tracheobronchial tumor, which affects the trachea even less often than bronchi. Because of its rarity, no large series has ever been published exclusively on tracheal mucoepidermoid tumors, and precise demographic data are lacking. Although it occurs in a wide age range, from childhood to the elderly, many cases are reported in children, and a predilection for the teenage
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and young adult population seems to be present, as is the case in its bronchial counterpart. In a large series of bronchial mucoepidermoid carcinomas, low-grade tumors comprised 90% of cases, and over half of them occurred in individuals younger than 30 years of age.51,52 High-grade tumors occurred in 10% of cases, 70% of these in individuals older than 30 years. Pathology. Grossly, this tumor typically grows as a polypoid tan-gray to pink endotracheal mass (Figures 3-29, 3-30 [Color Plate 4]). Invasion through the trachea into adventitia is seen in some high-grade tumors. Microscopically, mucoepidermoid carcinomas are characterized by a variable admixture of mucussecreting cells, squamous cells, and cells of intermediate type (Figures 3-31, 3-32 [Color Plate 4]).53 Mucinproducing cells are scattered individually, in clusters, or more commonly, arranged in glandular or cystic structures, which are intimately intermixed with sheets of intermediate or squamous cells. Intermediate cells are taken to be smaller than squamous cells, with scant cytoplasm and without any differentiation toward squamous or mucinous cells. Obvious features of squamous differentiation as squamous pearls, eddies, or dyskeratotic cells are often absent. Oncocytic and clear cell changes are described in bronchial counterparts. One case of oncocytic mucoepidermoid carcinoma has been reported in the trachea, in which sheets and nests of oncocytes comprised 75% of the tumor.54 The cells stained positive for phosphotungstic acid-hematoxylin (PTAH) and negative for PAS and neuroendocrine markers. Anaplastic features were absent and the tumor was classified as low grade. Regional lymph node metastasis was found. Clear cell change is not reported in tracheal tumors but could possibly occur as it does in bronchial tumors. Differential diagnosis includes squamous cell carcinoma, adenocarcinoma, other salivary gland-type tumors (like pleomorphic adenoma), carcinoid tumor, and mucous gland adenoma. The oncocytic variant must be differentiated from oncocytoma and carcinoid tumor. Adenosquamous carcinoma is a major differential diagnosis in the lungs (bronchial tree), but no case has been reported in the trachea. A grading system is not as well established as is in salivary gland mucoepidermoid carcinomas since there are no large follow-up studies. The grading system that follows is used for bronchial mucoepidermoid carcinomas and seems to correlate with tumor behavior and disease outcome in tracheal tumors as well.52 In low-grade mucoepidermoid carcinomas that comprise the majority, glands or cysts of mucinous cells predominate. The minor components are the solid sheets of intermediate and squamous cells, which show little mitotic activity, nuclear pleomorphism, or necrosis. High-grade tumors have sheets of intermediate and squamous cells with high mitotic rate (4 per 10 highpower fields on average), nuclear atypia, or cellular necrosis. Glandular structures are the minor component. Older literature is conflicting with respect to disease outcome and patient survival, partly due to the misclassification of this tumor with others as a “bronchial adenoma,” and partly due to the lack of a grading system. Hence, both excellent and very poor prognoses are reported, now attributable to low and high grades of tumors. The strong association between tumor grade and clinical course is now well established, with an excellent course in low-grade and a fatal course in most high-grade tumors.55,56
Malignant Melanoma Malignant melanoma of the skin can rarely metastasize to the trachea. Even less frequent is a malignant melanoma primary in the trachea, with only about 4 cases reported in the literature so far.57 An additional case of multiple tracheobronchial melanoma has also been published, in which the primary site could have been either the trachea or bronchi.58 Pathology. Grossly, most of these tumors have been polypoid, one of them with a long, narrow stalk (Figure 3-33 [Color Plate 5]).59 Another of the tumors had a flat subepithelial location.60 This flat melanoma had metastasized to the adjacent lymph node. Pulmonary metastasis was present in one case.59
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Microscopically, the tumor classically consists of large cells with atypical vesicular nuclei and prominent nucleoli. The cells are arranged in nests and sheets (Figure 3-34 [Color Plate 5]) and contain melanin. If no melanin is seen, one can prove melanocytic differentiation by immunohistochemical or ultrastructural analyses. Intraepithelial location of malignant melanoma suggests that the tumor has arisen at that site. Differential diagnosis is broad, and includes various tumor categories such as carcinomas, sarcomas, and lymphomas. The most important ones to differentiate in this location are large cell carcinoma and large cell neuroendocrine carcinoma.
Metastatic Tumors to the Trachea Secondary involvement of the trachea mostly occurs as direct invasion from adjacent organs, such as the larynx, thyroid (Figures 3-35, 3-36, 3-37 [Color Plate 5]), esophagus, or mediastinal structures, and is rarely due to metastasis (Figure 3-38 [Color Plate 5]). This is in contrast to the bronchial tree, which more commonly receives metastases from distant primaries. All published tracheal metastases consist of carcinomas and melanomas, and no sarcoma has been reported, although the latter has been recognized as a form of bronchial metastasis. Table 3-1 shows the demographic data and types of the 14 published cases of tracheal metastasis.21,22,61–71, Carcinomas of the breast and colon and cutaneous melanoma were the most common
Table 3-1 Reported Cases of Metastases to the Trachea Reference Number
Primary Tumor Site
Primary Tumor Type
Date
Age
Sex
61 62 63 22 64
1954 1965 1974 1975 1978
41 35 35 62 61
65
1980
66
M F F F M
Colon Colon Breast Endometrium Kidney
Adenocarcinoma Adenocarcinoma Medullary carcinoma Adenoacanthoma Renal cell carcinoma
6 years 3 years 5 years 11 years 6 years
61
F
Ovaries
Bilateral papillary cystadenocarcinoma
7 years
1980
57
F
Breast
6 years
66
1980
50
F
Breast
12 years
None
67
1981
28
M
2 years
None
68
1982
40
F
Melanoma
2 years
69 70
1987 1991
41 44
M F
Esthesioneuroblastoma Melanoma
1 year 2 months
21 71
1994 2001
73 56
M M
Skin (shoulder) Skin (upper arm) Nose Skin (breast) Colon Left lung
Invasive ductal carcinoma Invasive ductal carcinoma Melanoma
None ?Lung, ?Liver Lung Lung Femur, supraclavicular and mediastinal lymph nodes Pleura, axillary lymph nodes Lung
Adenocarcinoma Squamous cell carcinoma
13 months 2 years
Ribs, vertebral column None Pharynx, axillary lymph nodes Liver Pulmonary hilar lymph nodes
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metastatic tumors to the trachea. All patients had a history of primary cancer. Melanoma had the least time interval from diagnosis of primary cancer to recognition of tracheal metastases, which was 2 months in 1 of the cases and 2 years in 2 others.67,68,70 Concurrent or prior metastasis to the lung, lymph nodes, or bone had been present in most patients. Common symptoms at presentation were dyspnea, wheezing, cough, and hemoptysis. On bronchoscopy, most of the tumors showed a polypoid growth into the lumen, which was pedunculated in a minority of cases.69 Histologically, most of the tumors had a subepithelial location, some extending deep in the tracheal wall and even surrounding soft tissue. The overlying epithelium was hyperplastic or ulcerated. No melanocytic junctional activity was found in the overlying tracheal epithelium in melanoma cases, which was in keeping with the metastatic nature of the tumors. The morphology was similar to primary cancer in all cases. The fact that the primary tumor had been identified in all reported cases eases the differential diagnosis with primary tracheal tumors. Nevertheless, when dealing with primary malignant epithelial tumors of the trachea, especially adenocarcinomas, a metastatic disease should be considered in the differential diagnosis, particularly in cases in which sufficient clinical history is not provided. The prognosis depends on a multitude of factors, including primary tumor site and type, tumor stage, and respiratory function following tracheal luminal narrowing.
References 1. 2.
3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13. 14. 15.
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Kaufman G, Klopstock R. Papillomatosis of the respiratory tract. Am Rev Resp Med 1963;88:839–46. Popper HH, Wirsberger G, Juttner-Smoll FM, et al. The predictive value of human papilloma virus (HPV) typing in the prognosis of bronchial squamous cell papillomas. Histopathology 1992;21:323–30. Di Mario AF, Montenegro H, Payne CB Jr, Kwon KH. Papillomas of the tracheobronchial tree with malignant degeneration. Chest 1978;74:464–5. Runckel D, Kessler S. Bronchogenic squamous carcinoma in nonirradiated juvenile laryngotracheal papillomatosis. Am J Surg Pathol 1980;4:293–6. Fechner RE, Fitz-Hugh GS. Invasive tracheal papillomatosis. Am J Surg Pathol 1980;4:79–80. Dail D. Uncommon tumors. In: Dail DH, Hammar SP, editors. Pulmonary pathology. 2nd ed. New York: Springer-Verlag; 1994. p. 1299–300. Hajdu SI, Huvas AG, Goodner JT, et al. Carcinoma of the trachea, clinicopathologic study of 41 cases. Cancer 1970;25:1448–56. Salm R. Primary carcinoma of the trachea. A review. Br J Chest 1964;58:61–72. Grillo HC, Mathisen DJ. Primary tracheal tumors: treatment and results. Ann Thorac Surg 1990;49:69–77. Houston HE, Payne WS, Harrison EG, Olsen AN. Primary cancers of the trachea. Arch Surg 1969;99:132–40. Bennets FE. Tracheal tumors. Postgrad Med J 1969; 45:446–54. Theegarten D, Freitag L. Scar carcinoma of the trachea after tracheostomy. Case report and review of the literature. Respiration 1993;66:250–3. Theegarten D, Philippou S, Freitag L. Tracheal carcinoma and mixed pneumoconiosis. A causal relationship? Respiration 1995;62:49–52. Hatta T, Tsubota N, Matsubara M, et al. Two cases of metachronous cancer of the lung and trachea. Kyoba Geta 1988;41:422–5. Shan L, Nakamura Y, Nakamura M, et al. Synchronous and metachronous multicentric squamous cell carcinoma in the upper aerodigestive tract. Pathol Intern 1997;47:68–72. Saltarelli MG, Fleming MW, Wenig BM, et al. Primary
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basaloid squamous cell carcinoma of the trachea. Am J Clin Pathol 1995;104:594–8. Thornley PE, Howtson SR. Primary carcinoma of the trachea: mixed squamous and oat cell type. Thorax 1980;35:72–3. Gelder CM, Hetzel MR. Primary tracheal tumours: a national survey. Thorax 1993;48:688–92. Dalby JE, Jones MN. Primary malignant growths of the trachea. Acta Otolaryngol 1961;53:12–20. Zirkin HJ, Tovi F. Tracheal carcinoma presenting as a thyroid tumor. J Surg Oncol 1984;26:268–71. Conti JA, Kemeney N, Klimstra D, et al. Colon carcinoma metastatic to the trachea. Report of a case and a review of the literature. Am J Clin Oncol 1994; 17:227–9. Zerner J. Metastatic carcinoma (endometrial adenoacanthoma) to the trachea. Report of a successful resection and primary anastomosis. J Thorac Cardiovasc Surg 1975; 70:139–42. Mori S, Shinoda M, Hatooka S, et al. A carcinoma arising from benign pleomorphic adenoma of the trachea. Kyobu Geka 1997;50:602–5. Rotenberg D, Lawson VG, van Nostrand AW. Thyroid carcinoma presenting as a tracheal tumor. Case report and literature review with reflections on pathogenesis. J Otolaryngol 1979;8:401–10. Sobin L, Yesner R. Histological typing of lung tumors. International histological classification of tumors. Vol 1. 2nd ed. Geneva: World Health Organization; 1981. Downey RJ, Deschamps C, Asakura S, et al. Large cell carcinoma of the lung: results of surgical treatment. Lung Cancer 1994;11 Suppl:153. Fields JN, Riguad G, Emami BN. Primary tumors of the trachea. Results of radiation therapy. Cancer 1989;15:2429–33. Fishback N, Travis W, Moran C, et al. Pleomorphic (spindle/giant cell) carcinoma of the lung: a clinicopathological study of 78 cases. Cancer 1994;73:2936–45. Onizuka M, Doi M, Mitsui K, et al. Undifferentiated carcinoma with prominent lymphocytic infiltration (socalled lymphoepithelioma) in the trachea. Chest 1990;98:236–7
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Marigil MA, Pardo-Mindan FJ, Aliaga L, et al. Primary carcinoma of the trachea: combined small cell, squamous cell and giant cell carcinoma. Report of a case and review of the literature. Pathologica 1986; 78:99–105. Passler C, Scheuba C, Prager G, et al. Anaplastic (undifferentiated) thyroid carcinoma (ATC). A retrospective analysis. Langenbecks Arch Surg 1999; 384:284–93. Arrigoni MG, Woolner LB, Bernatz PE. Atypical carcinoid tumors of the lung. J Thorac Cardiovasc Surg 1972;64:413–21. Hulka GF, Rothchild MA, Warner BW, Bove KE. Carcinoid tumor of the trachea in a pediatric patient. Otolaryngol Head Neck Surg 1996;114:822–5. McCaughan BC, Martini N, Bains MS. Bronchial carcinoids. Review of 124 cases. J Thorac Cardiovasc Surg 1985;89:8–17. Kharchenko VP, Galil-ogly GA, Kuz’min IV, et al. Melanocytic carcinoid of the trachea. Arch Pathol 1992;54:38–41. Travis WD, Rush W, Flieder DW, et al. Survival analysis of 200 pulmonary neuroendocrine tumors with clarification of criteria for atypical carcinoid and its separation from typical carcinoid. Am J Surg Pathol 1998;22:934–44. Colby TV, Koss MN, Travis WD. Carcinoid and other neuroendocrine tumors. In: Atlas of tumor pathology. Tumors of the lower respiratory tract. Washington (DC): Armed Forces Institute of Pathology; 1995. p. 287–317. Tartour E, Caillou B, Tenebaum F, et al. Neuroendocrine tumor of the trachea of the intermediate type. Value of its individualization. Presse Med 1992;21:1905–8. Colby TV, Koss MN, Travis WD. Small cell carcinoma and large cell neuroendocrine carcinoma. In: Atlas of tumor pathology. Tumors of the lower respiratory tract. Washington (DC): Armed Forces Institute of Pathology; 1995. p. 248–57. Rostom AY, Morgan RL. Results of treating primary tumors of the trachea by irradiation. Thorax 1978;33:387–93. van Heyningen C, Green AR, MacFarlane IA, Burrow CT. Oncogenic hypophosphatemia and ectopic corticotrophin secretion due to oat cell carcinoma of the trachea. J Clin Pathol 1994;47:82–2. Ma CK, Fine G, Lewis J, Lee MW. Benign mixed tumor of the trachea. Cancer 1979;44:2260–6. Ishida T, Kamachi M, Hanada T, et al. Mucous gland adenoma of the trachea resected with an endoscopic neodymium:yttrium aluminum garnet laser. Intern Med 1996;35:890–3. Ferguson CJ, Cleeland JA. Mucous gland adenoma of the trachea: case report and literature review. J Thorac Cardiovasc Surg 1988;95:347–50. Azar T, Abdulkarim FW, Tacher HM. Adenoid cystic carcinoma of the trachea. Laryngoscope 1998;108: 1297–300. Maziak DE, Todd TR, Keshavjee SH, et al. Adenoid cystic carcinoma of the airway: thirty-two year experience. J Thorac Cardiovasc Surg 1996;112:1522–32. Na DG, Han MH, Kim KH, et al. Primary adenoid cystic carcinoma of the cervical trachea mimicking thyroid tumor: CT evaluation. J Comput Assist Tumor 1995;19:559–63. Nomori H, Kaseda S, Kobayashi K, et al. Adenoid cystic carcinoma of the trachea and main-stem bronchus. A clinical, histopathologic, and immunohistochemical study. J Thorac Cardiovasc Surg 1988;96:271–7.
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57. 58. 59. 60. 61. 62.
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Umeno H, Miyajina Y, Mori K, Nakashima T. Clinicopathological study of 54 cases of adenoid cystic carcinoma in the head and neck. Nippon Jibiinkoka Gakkai Kaiho 1997;100:1442–9. Smetana HF, Iverson L, Swan LL. Bronchogenic carcinoma. Analysis of 100 autopsy cases. Milit Surg 1952;3:335. Liebow AA. Tumors of the lower respiratory tract. In: Atlas of tumor pathology. Tumors of the lower respiratory tract. Washington (DC): Armed Forces Institute of Pathology; 1975. p. 26–53. Yousem SA, Hochholzer L. Mucoepidermoid tumors of the lung. Cancer 1987;60:1346–62. Schanmugarten K. Histological typing of tumors of the upper respiratory tract and ear. Berlin: World Health Organization; 1991. p. 35–6. Lopez-Terrada D, Bloom MGK, Cagle PT, et al. Oncocytic mucoepidermoid carcinoma of the trachea. Arch Pathol Lab Med 1999;123:635–7. Heitmiller RF, Mathisen DJ, Ferry JA, et al. Mucoepidermoid lung tumors. Ann Thorac Surg 1989;47:394–9. Leonardi HK, Jung-Legg Y, Legg MA, Neptune WB. Tracheobronchial mucoepidermoid carcinoma. Clinicopathological features and results of treatment. J Thorac Cardiovasc Surg 1978;76:431–8. Duarte IG, Gal AA, Mansour KA. Primary malignant melanoma of the trachea. Ann Thorac Surg 1998; 65:559–60 Kharchenko VP. Primary melanoma of the respiratory tract. Arch Pathol 1998;60:38–41. Eri Z, Stanic J, Stanic M. Primary malignant melanoma of the trachea. Plucne Bolesti 1991;43:75–7. Mori K, Cho H, Som M. Primary “flat” melanoma of the trachea. J Pathol 1977;121:101–5. Divertie MB, Schmidt HW. Tracheal obstruction from metastatic carcinoma of the colon. Report of a case. Mayo Clin Proc 1954;29:403–5. Yeh TJ, Batayias G, Peters H, Ellison RG. Metastatic carcinoma of the trachea. Report of a case of palliation by resection and Marlex graft. J Thorac Cardiovasc Surg 1965;49:886–92. Garces M, Tsai E, Marsan RE. Endotracheal metastasis. Chest 1974;65:350–1. MacMohan H, O’Connel DJ, Cimochowski GE. Pedunculated endotracheal metastasis. Am J Radiol 1978; 131:713–4. Westerman DE, Urbanetti JS, Rudders RA, Fanburg BL. Metastatic endotracheal tumor from ovarian carcinoma. Chest 1980;77:798–800. Baumgartner WA, Mark JB. Metastatic malignancies from distant sites to the tracheobronchial tree. J Thorac Cardiovasc Surg 1980;79:499–503. Andrews AH Jr, Cardarelli DD. Carbon dioxide laser treatment of metastatic melanoma of the trachea and bronchi. Ann Otol 1981;90:310–1. Wicks CM, Pos RH, Emerson GL, Kallay MC. Malignant melanoma metastatic to trachea. Clin Note Resp Dis 1982;21:14–6. Franklin D, Miller RH, Kim Bloom MG, et al. Esthesioneuroblastoma metastatic to the trachea. Head Neck Surg 1987;10:102–6. Nicolai P, Peretti G, Capiello J, et al. Melanoma metastatic to the trachea and nasal cavity: description of a case and review of the literature. Acta Otorhinolaryngol Ital 1991;11:85–92. Ishiyama T, Aoyama T, Hirahara H, et al. Successful resection of endotracheal metastatic lung cancer using percutaneous cardiopulmonary support system: a case report. Kyobu Geka 2001;54:19–23.
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3B MESENCHYMAL TUMORS OF THE TRACHEA Javad Beheshti, MD Eugene J. Mark, MD
Fibroblastic and Fibrohistiocytic Tumors Muscle Tumors Lipomatous Tumors Cartilaginous Tumors Vascular Tumors Nerve Sheath Tumors
Synovial Sarcoma Hamartoma Paraganglioma (Chemodectoma) Glomus Tumor Lymphomas
Mesenchymal neoplasms, also generally termed “soft tissue tumors,” affect the trachea like any other organ or tissue of the body, because the trachea is surrounded by mesenchyme and has mesenchymal cells in its wall and mucosa. These include fibroblasts in the lamina propria, smooth muscle cells in the posterior membranous septum, and chondrocytes in the cartilage. Because of their rarity, mesenchymal lesions are rarely suspected, and the supposition is that a tracheal tumor is epithelial until proven otherwise. The distinction between benign and malignant mesenchymal lesions is in general less distinct than in epithelial lesions. In contrast to epithelial lesions, a greater proportion of mesenchymal lesions of the trachea are benign, or are of such low-grade malignancy that metastasis is unusual. Even malignant lesions, by virtue of their location and circ*mscription, are still generally amenable to resection and cure. Sampling problems tend to be greater in mesenchymal than in epithelial lesions, so an initial small biopsy is often only a signpost to the final diagnosis established after resection of the specimen. Lymphomas are rare in the trachea and are included in this section.
Fibroblastic and Fibrohistiocytic Tumors Fibroblastic and fibrohistiocytic tumors are perhaps the least delineated and clarified area in tracheal tumor pathology. Different names have been given to pathologically identical or very similar lesions, and different histopathologies have been lumped under single names. The confusion between fibroma, fibromatosis, and fibrosarcoma on the one hand, and between benign and malignant fibrous histiocytomas on the other, exemplify this situation. Inflammatory pseudotumor has also been added. It is our belief that the pathologic entities in this group generally should be equated to these lesions in general mesenchymal tumor pathology, unless meaningful data prove otherwise in the future.
Pathology of Tracheal Tumors
Fibroma Fibroma is found to be one of the most common benign tracheal tumors reported in the older literature, but it is rarely reported in recent publications.1,2 We believe that most of the reported cases likely represented fibromatosis, low-grade fibrosarcoma, benign fibrous histiocytoma, inflammatory myofibroblastic tumor (inflammatory pseudotumor), or granulation and reactive fibroblastic tissues. Tracheal lesions consisting of fascicles of bland fibroblastic cells should be classified in one of the above-mentioned entities.
Fibromatosis Fibromatosis, as proposed by Stout, consists of a broad group of benign fibrous proliferations of similar microscopic appearance that are intermediate in their biological behavior between benign fibrous lesions and fibrosarcoma.3 Like fibrosarcoma, they are characterized by infiltrative growth and a tendency toward recurrence, but unlike this tumor, they never metastasize.4 Fibromatosis of the trachea is less common than mediastinal fibromatosis invading the trachea. It probably represents most of the previously reported cases of “tracheal fibroma” and is more common in children. This lesion usually presents as a subepithelial nodule, and is a benign proliferation of bland regular fibroblasts arranged in short fascicles. The margins are infiltrative. The overlying epithelium may show hyperplasia or squamous metaplasia.5 The major differential diagnosis is low-grade fibrosarcoma. Fibrosarcoma is usually more cellular, contains long fascicles of more pleomorphic fibroblasts, and exhibits more mitotic activity. Benign fibrous histiocytoma is distinguishable by the presence of histiocytes and inflammatory cells. Inflammatory myofibroblastic tumor (inflammatory pseudotumor) is separated by the presence of inflammatory cells and myofibroblastic differentiation in the proliferative cells. Because of its infiltrative growth, tracheal fibromatosis commonly recurs after incomplete excision. Prognosis is good after complete resection.
Fibrosarcoma Very few cases of primary tracheal fibrosarcoma have been reported in the literature, and most have been reported in children.6,7 It occurs as a neoplastic proliferation of fibroblasts, producing a mass lesion that protrudes into the tracheal lumen. Histologically, tracheal fibrosarcoma consists of spindle cells with somewhat atypical oval nuclei, arranged in long fascicles and in a herringbone pattern (Figure 3-39 [Color Plate 5]). Mitotic figures are seen. Differential diagnosis includes fibromatosis, benign and malignant fibrous histiocytomas, inflammatory myofibroblastic tumor, and other spindle cell tumors. Treatment is the same as for fibromatosis, which involves complete excision. Although primary tracheal fibrosarcoma has the potential for metastasis, this did not occur in the few cases reported in the trachea, and prognosis has generally been good.
Benign Fibrous Histiocytoma This tumor usually presents as a polypoid mass, protruding into the tracheal lumen. Microscopically, it consists of a poorly circ*mscribed proliferation of elongated fibroblastic and polygonal histiocytic cells, intermixed with collagen fibers, with occasional multinucleated giant cells and dispersed lymphoid cells. A vague fascicular arrangement or a storiform pattern is often seen. Foam cells and siderophages are less frequently present. Cellular pleomorphism is minimal, and mitotic figures are scant. A few cases of benign fibrous histiocytoma have been reported in the literature, mostly in children and young adults.8–10 Cases of this tumor are classified in some publications under the general term “fibrous
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histiocytoma” or “malignant fibrous histiocytoma,” partly because of its infiltrative nature and recurrence after excision. Differential diagnosis includes fibromatosis, inflammatory pseudotumor, neurofibroma, fibrosarcoma, and hemangiopericytoma. The distinction from malignant fibrous histiocytoma is often obvious, with the latter showing marked cellular pleomorphism, high mitotic activity, and foci of necrosis. Benign fibrous histiocytoma is infiltrative at the margins and may recur after incomplete excision. The prognosis is good after proper excision.
Malignant Fibrous Histiocytoma Very few cases of malignant fibrous histiocytoma (MFH) have been reported in the trachea. One case of postirradiation MFH was reported to have occurred 11 years after radiotherapy to the neck for thyroid papillary carcinoma.11 It involved almost the entire length of the tracheal wall, and caused death in about 3 months. A co-occurrence of tracheal MFH with thymic carcinoma, producing separate lesions in the mediastinum, has also been reported.12 Histology shows pleomorphic spindle cells arranged in storiform and fascicular patterns. Pleomorphic giant cells are dispersed and inflammatory cells may be seen. Mitotic figures are frequent and foci of necrosis may be present. Differential diagnosis is mainly with other spindle cell sarcomas.
Muscle Tumors Leiomyoma Leiomyoma is a benign smooth muscle tumor that is far less common in the trachea than in bronchi and lung parenchyma. Approximately 21 cases of solitary leiomyoma of the trachea have been reported in the English and Japanese literatures. Multiple leiomyoma of the trachea, as well as other organs such as the esophagus and female genital tract, is associated with Alport’s syndrome.13 A case of esophageal leiomyomatosis involving the trachea has also been reported.14 Biology. In one series of 16 patients, the tumors appeared approximately equally in males and females.15 The age range was 15 to 72 years, with two-thirds of the patients being above 40 years. The mean age was 49 years in another series of 20 cases.16 Pathology. More than one-half of the cases have been in the lower third of the trachea, and most have involved the posterior wall, where smooth muscle is most abundant. The tumors have been measured between 1 and 2.5 cm. The tumors usually protrude into the lumen as a nodule (Figure 3-40 [Color Plate 5]). Two cases were pedunculated. Microscopically, leiomyoma consists of monomorphic spindle cells with blunt-ended nuclei, arranged in interlacing fascicles and storiform patterns. Significant mitotic activity and necrosis are absent (Figure 3-41 [Color Plate 6]). Differential diagnosis includes leiomyosarcoma, as well as other spindle cell tumors such as hemangiopericytoma and fibrous histiocytoma.
Leiomyosarcoma This malignant smooth muscle tumor is also rare in the trachea, with only 18 cases reported in the literature so far. It produces an intraluminal mass, which was pedunculated in 2 cases.17,18 Most of the cases in one series were in the upper third of the trachea.17
Pathology of Tracheal Tumors
Microscopically, the tumor consists of interlacing fascicles of spindle cells that are similar to leiomyoma. Distinguishing features from leiomyoma are cellular anaplasia, sometimes with the presence of uni- or multinucleated giant cells, high mitotic activity, and necrosis. The most helpful feature is the mitotic rate, which is more than 1 to 2 per 10 high-power fields, although in most cases, it is more than that; some believe it must be more than 5 per 10 high-power fields.17–19 Differential diagnosis includes other spindle cell sarcomas and sarcomatoid carcinomas.
Rhabdomyosarcoma Rhabdomyosarcoma is a malignant mesenchymal neoplasm with differentiation toward skeletal muscle tissue. The tumor is generally seen in children. Two cases of this tumor in the trachea have been published in the Englishlanguage literature. One was in a 65-year-old man, with a polypoid mass in the tracheal lumen causing symptoms.20 Histologically, the mass was inner to the fibrocartilaginous layer and covered by overlying epithelium. It consisted of anaplastic cells with large hyperchromatic nuclei, and abundant brightly eosinophilic cytoplasm, some showing cross-striations. The tumor was classified as a pleomorphic rhabdomyosarcoma. The second case occurred in a 12-year-old girl, also with an intratracheal polypoid mass. Histologically, this mass consisted of rather small, round, and spindled hyperchromatic cells, and was diagnosed as an embryonal rhabdomyosarcoma.21 More common is the mediastinal rhabdomyosarcoma with pressure on the trachea.22
Lipomatous Tumors Lipoma-Liposarcoma Lipoma is a benign mesenchymal neoplasm of fat and is most common in the subcutis. In the usual type, it resembles mature fat, surrounded by a delicate capsule. It is exceedingly rare in the trachea, with only approximately 10 cases reported in the literature. Lipomas produce a polypoid mass covered by respiratory epithelium. One of the reported cases did not produce any symptoms and was found on autopsy,23 whereas 2 other cases caused airway obstruction.23–25 Microscopically, tracheal lipomas are composed of lobules of mature adipocytes, separated by delicate fibrous bands. One case of a well-differentiated liposarcoma in the trachea occurred in a 76-year-old man.26 It produced a 1 cm polyp, which was histologically composed of mature adipocytes with foci of atypicality. Recurrence or metastasis did not occur in 12 months of follow-up. The current trend is to classify these tumors as “atypical lipoma,” because although recurrence occurs, they do not metastasize.27 Since the recurrence rate is higher in retroperitoneal tumors, some prefer the term “well-differentiated liposarcoma” in that location. The major differential diagnosis is a hamartoma. In a hamartoma, fat tissue may be prominent, but the presence of cartilage and epithelium-lined clefts help to make the diagnosis.
Cartilaginous Tumors Chondroma Chondroma is a benign tumor composed of cartilage and can rarely arise in the trachea. Approximately 10 cases of chondroma are reported in the trachea.28,29 Grossly, it forms a hard bosselated mass protruding into the lumen. Microscopically, it is a relatively hypocellular tumor, composed of chondrocytes that sit in lacunae of hyaline cartilage. The chondrocytes may be somewhat hyperchromatic and pleomorphic. Binucleation is exceptional. Foci of ossification may be seen. Differential diagnosis includes, most importantly, chondrosarcoma, as well as hamartoma and tracheopathia osteoplastica. Hamartomas are predominantly composed of hyaline cartilage, but presence of fat, occasional smooth muscle fibers, and epithelium-lined clefts occur. Tracheopathia osteoplastica is multiple and usually distinguishable on bronchoscopy.
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Chondroblastoma Chondroblastoma is a benign tumor of bone and rarely of soft tissue. It usually affects patients in the second decade of life. We have seen 1 case of chondroblastoma occurring in the trachea. To our knowledge, there is no comparable case in the literature. Our patient was a 20-year-old male with a soft and friable, tan-yellow mass, protruding and partially obstructing the lumen. Grossly, there was hemorrhage and calcification in the tumor (Figure 3-42 [Color Plate 6]). Microscopically, the tumor was mainly in the submucosa. There was focal erosion of cartilage as well as extension to peritracheal adventitia. Sheets of mononuclear cells with the features of chondroblasts were present, including round or oval nuclei and pale eosinophilic cytoplasm. Nuclear grooves were seen in some cells (Figures 3-43, 3-44 [Color Plate 6]). Occasional multinucleated giant cells were present. A chondroid matrix and mature cartilage were seen. The overlying epithelium showed reactive squamous metaplasia. The tumor was completely excised and did not recur, which is also usually the case with skeletal chondroblastomas.
Chondrosarcoma Chondrosarcoma, the malignant counterpart of chondroma, is also rare in the trachea, with only 11 cases reported in the literature.29,30 The age range of the patients was 32 to 87 years with an average of 65 years. The male to female ratio was 8:1.30 Similar to chondroma, chondrosarcoma produces a hard intraluminal projection, which may be pedunculated. Tracheal obstruction may occur. Microscopically, the tumor consists of cells with hyperchromatic and sometimes pleomorphic nuclei, and occasionally binucleated cells, some containing prominent nucleoli. The cellularity is increased compared to that of chondroma. The matrix is chondroid and may contain areas of myxoid change. Because mitoses are few and inconsistent, one cannot rely on mitotic activity to separate benign and malignant cartilaginous tumors in the same way as one does for the differentiation of benign from malignant smooth muscle tumors. The major differential diagnosis is chondroma, which is often not an easy distinction. The criteria for malignancy are given above, but since chondrosarcomas of the larynx and trachea are usually low-grade and slow-growing, the cytologic changes may be subtle. Malignant nature of the tumor has been determined only after recurrence in some published cases. Some propose the term “cartilaginous tumor” for both chondroma and chondrosarcoma of this site because of the difficulty in separation. The treatment of both tumors is the same; that is, excision. After resectional surgery, chondromas and chondrosarcomas infrequently recur.
Vascular Tumors Hemangioma Capillary hemangioma affects the larynx and trachea of children far more commonly than it does adults, and because of the narrower airway lumen in this younger age group, it becomes symptomatic early in growth and causes obstructive symptoms such as stridor and dyspnea.31 Most of the cases are well circ*mscribed, but some diffusely infiltrate the wall and cause narrowing over a segment, or grow into adjacent mediastinal tissues.32 Conversely, a few cases of mediastinal hemangioma have been reported with infiltrative growth through the tracheal wall into the lumen.33 Also published are cases of mediastinal and cervical hemangioma with compression of, but not infiltration into the trachea, causing symptoms.34,35 Histologically, capillary hemangiomas consist of closely packed capillary-sized blood vessels with marked endothelial proliferation, often with recognizable lobular architecture.
Pathology of Tracheal Tumors
A major differential diagnosis is granulation tissue, which represents reparative and inflamed connective tissue rich in capillaries, formed in response to trauma or endotracheal intubation.36 The distinction can be made by the presence of lobularity in hemangiomas, the absence of inflammation in deep areas in hemangiomas, and a history of trauma or endotracheal intubation in cases with granulation tissue. Recurrence has been reported after incomplete excision.33
Kaposi’s Sarcoma Kaposi’s sarcoma is a malignant vascular tumor seen in immunocompromised patients, particularly those affected with human immunodeficiency virus (HIV). When the trachea is involved, it is often in a disseminated form of disease with involvement of other sites such as bronchi, the larynx, palate, or distant organs like the skin.37,38 Biology. Predisposition to multifocal Kaposi’s sarcoma may also develop in drug-induced immunosuppression, as occurred in the trachea and bronchi of a lung transplant patient receiving immunosuppressive therapy.39 It is rarely seen in patients without immunosuppression.40 Solitary lesions in the trachea have also been reported.2 Hemorrhage into the trachea may dominate the clinical presentation. Pathology. The endoscopic appearance may be suspicious for diagnosis without biopsy. Histologically, Kaposi’s sarcoma is composed of proliferating spindle cells with slit-like vascular channels, extravasated red blood cells, and inflammatory cells. Cytoplasmic hyaline bodies may be seen in the cytoplasm of the malignant cells. Ectatic blood vessels may be seen in the surrounding tissue (Figures 3-45, 3-46, 3-47 [Color Plate 6]). Differential diagnosis includes principally granulation tissue and spindle cell sarcomas.
Hemangiopericytoma Hemangiopericytoma is a tumor of vascular pericytes, 4 cases of which have been reported in the trachea. Hemangiopericytoma is a potentially malignant tumor and its clinical behavior is not always possible to predict from morphology alone. Microscopically, the tumor consists of closely-packed round to oval cells with scant cytoplasm, arranged around thin-walled “staghorn” blood vessels (Figure 3-48 [Color Plate 6]). A mitotic rate of generally more than 4 per 10 high-power fields is one indicator of malignant behavior, as was present in a published case, which recurred after primary excision, with massive infiltration of peritracheal cervical and mediastinal soft tissues.41 Of the 4 cases reported, 2 had aggressive behavior and recurrence, so clinical follow-up is important. One case of hemangiopericytoma arising in the mediastinum and compressing the trachea, and another case arising in the thyroid gland with invasion of the larynx, have been reported.42
Nerve Sheath Tumors This category of tumors includes neurofibroma, schwannoma, and malignant peripheral nerve sheath tumor. These tumors are among the common tumors in the mediastinum, especially the posterior compartment. They may cause a pressure effect on mediastinal structures including the trachea. Neurofibromas and schwannomas are infrequent as endotracheal tumors. No case of malignant peripheral nerve sheath tumor has been reported in the trachea, but tracheal involvement has occurred in this tumor originating from the vagus nerve.43
Neurofibroma Neurofibroma is a benign tumor of Schwann cell origin and fibroblasts. The axons present within the tumor are continuous with adjacent nerves.
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Biology. Neurofibromas may occur as a solitary tumor in the trachea, or as part of von Recklinghausen’s neurofibromatosis.44 The two conditions occur with approximately equal frequency.45 One case was reported as multiple neurofibromas of the trachea, bronchi, and esophageal wall.46 The presence of multiple neurofibromas suggests von Recklinghausen’s neurofibromatosis. Pathology. Grossly, the tumor presents as an intraluminal polypoid mass, which is sessile or pedunculated. Microscopically, the tumor consists of slender spindle cells, sometimes showing wavy nuclei, admixed with fibroblasts in a haphazard fashion. Neurofibromas may be cellular or contain myxoid areas. Mitotic activity is not present. The tumor margins are often ill defined. Differential diagnosis includes schwannoma, fibrous histiocytoma, and other spindle cell tumors.
Schwannoma (Neurilemoma) Shwannoma is another tumor of Schwann cell origin with rare occurrence in the trachea. Biology. Unlike neurofibromas, schwannomas occur as solitary lesions in the trachea, and are not associated with von Recklinghausen’s neurofibromatosis. In a series of 12 patients, the age range was 6 to 71 years, with almost equal occurrence in males and females.47 Pathology. Schwannomas occur most frequently in the lower third of the trachea, followed by the upper and middle thirds.47,48 Grossly, schwannomas are encapsulated tumors which may be pedunculated or sessile, and in rare cases, are dumbbell-shaped with an extratracheal component (Figure 3-49 [Color Plate 7]). Schwannomas of the vagus nerve, although extratracheal, may involve the trachea.49 Microscopically, schwannoma consists of interlacing fascicles of spindle cells with hypercellular (Antoni type A) and hypocellular (Antoni type B) areas (Figures 3-50 [Color Plate 7]). Nuclear palisade (Verocay bodies) may be seen in hypercellular regions (Figures 3-51 [Color Plate 7]). Bizarre degenerative cells may develop as the tumor ages in a phenomenon termed “ancient schwannoma”. Differential diagnosis includes neurofibroma, fibrosarcoma, fibrous histiocytoma, and other spindle cell tumors. Malignant schwannoma occurs (Figure 3-52 [Color Plate 7]).
Granular Cell Tumor Granular cell tumor is a tumor of Schwann cell origin and commonly occurs in the tongue, but it has also occurred throughout the body. The trachea is less commonly involved than the larynx and bronchi. Approximately 40 cases have been reported in the trachea. Biology. In the largest published review of this tumor in the trachea, which comprised 30 cases, there was a female to male ratio of 6.5:1.50 Blacks were more commonly affected than whites. The age range was between 6 and 56 years with a peak incidence in the fourth decade. In another published series of tracheal tumors in children, granular cell tumor followed after hemangioma as one of the most common benign tumors in this age group.51 Pathology. The tumor size is usually between 0.5 to 6 cm (Figure 3-53 [Color Plate 7]). The majority arise from the cervical trachea.50 Twenty percent of the cases were multiple. Although most of the tumors (73%) were intraluminal lesions, 17% of cases grew extraluminally into surrounding tissues. This extraluminal
Pathology of Tracheal Tumors
growth and potential to penetrate adjacent tissues or organs can cause diagnostic problems, with the tumor presenting as a neck mass, or thyroid or parathyroid nodule.52–54 Microscopically, the tumor consists of round, oval, or polyhedral cells with granular eosinophilic cytoplasm, and small and rather central nuclei, occasionally with conspicuous nucleoli (Figure 3-54 [Color Plate 7]). The cytoplasmic granules stain for PAS antigen and neuron specific enolase. Squamous metaplasia with pseudoepitheliomatous hyperplasia is seen in the overlying epithelium. The cytoplasmic granules have characteristic features on electron microscopy. These are membrane-bound granules, also called “secondary lysosomes,” the larger ones having a lamellated structure and the smaller ones a granular content. Malignant granular cell tumors are extremely rare, not exceeding 1 to 2% of all granular cell tumors. They have pleomorphic nuclei with increased mitotic activity and foci of necrosis. Only 1 case has been reported near the trachea, and that was in the retrotracheal space with multiple nodules in both lungs (presumably metastases). Differential diagnosis includes oncocytoma, and neoplasms with oncocytic (Hurthle cell) change, oncocytic carcinoid tumors, and metastatic carcinomas with oncocytic change. The positive S-100 staining, negative neuroendocrine and keratin markers distinguish this tumor from carcinoid tumors and carcinomas. Hurthle cell variants of thyroid adenomas and oncocytomas are important because cases of extraluminal growth may present as thyroid nodules. In fine needle aspiration of such a thyroid nodule, the cells contain large, granular eosinophilic cytoplasm resembling Hurthle cells, and may cause misinterpretation.53 The distinction is by electron microscopy, which shows secondary lysosome rather than mitochondria of Hurthle cells. In malacoplakia, the cells are PAS negative with occasional MichaelisGutmann bodies.
Synovial Sarcoma Synovial sarcoma is primarily a sarcoma of the extremities. It has been reported in the head and neck region on rare occasion and can occur in the pleura and the lung.55 Only 1 case of this tumor has been reported in the trachea.56 The patient was a 20-year-old asthmatic white male, who had worsening of respiratory symptoms necessitating bronchoscopy. On bronchoscopy, a smooth pale intratracheal mass was seen. Gross examination of the resected specimen showed extension to paratracheal tissue without fat invasion. The tumor was well circ*mscribed. No lymph node metastases were detected. Microscopically, the tumor had a biphasic growth pattern, composed mostly of monomorphic spindle cells, but it also showed numerous foci of cuboidal cells forming pseudoglandular clusters. Immunohistochemistry showed vimentin positivity in the spindle cells and low molecular weight cytokeratin and carcinoembryonic antigen positivity in the epithelial component. Differential diagnosis is mainly with spindle cell tumors, most of which are separated by the lack of a biphasic pattern. Even if this aspect is not evident on routine histology of synovial sarcomas, it could be highlighted by immunohistochemical studies, as described in this case. The biphasic epithelioid-spindle cell tumors are generally in the differential diagnosis of synovial sarcoma. Within this category in pulmonary pathology, are mixed mesotheliomas, which must be distinguished from primary synovial sarcoma of the pleura, a tumor more recently demonstrated in this location. Sarcomatoid carcinoma of the lung and pulmonary blastoma are also in this category and must be separated from primary or metastatic synovial sarcoma. However, the mentioned biphasic tumors are not reported in tracheae.
Hamartoma Hamartoma, also called “cartilaginous hamartoma,” or less often “benign mesenchymoma,” is one of the common benign tumors of the lung, occurring mostly in lung parenchyma, with a minority occurring as bronchial
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lesions. Approximately 10 cases have been reported in the trachea, including cases in adults and children.57 Most of the cases had a polypoid appearance on endoscopy, causing obstruction of the lumen. One extraluminal hamartoma in a child presented as a neck mass attached to the trachea.58 Two cases were associated with pulmonary hamartomas, one with a similar lesion in lung parenchyma, and the other with multiple tumors in bronchi and lung parenchyma.59,60 Microscopically, hamartomas consist of lobules of mature hyaline cartilage and other mesenchymal elements, including undifferentiated mesenchymal tissue, fat, smooth muscle, with entrapped epitheliumlined clefts. The major differential diagnoses are chondroma and tracheopathia osteoplastica. Chondroma, by definition, does not contain mesenchymal elements other than cartilage, and does not have epithelial elements. Tracheopathia osteoplastica usually produces multiple nodules, which carpet the mucosa and consist of columns of bone and cartilage forming struts internal to the tracheal rings. Mediastinal teratoma should be considered in the differential diagnosis of extraluminal tracheal hamartomas.
Paraganglioma (Chemodectoma) Paraganglioma is a potentially malignant, usually low-grade tumor of the extra-adrenal paraganglia. Only 7 cases have been reported as primary in the trachea.61,62 It is characterized by organoid nests of epithelioid cells (zellballen pattern) separated by delicate vascularized connective tissue. The cells contain round or oval vesicular or hyperchromatic nuclei and light eosinophilic cytoplasms that are sometimes vacuolated. Neurosecretory granules are demonstrable in the cytoplasm by electron microscopy. Differential diagnosis includes carcinoid and glomus tumors. Carcinoid tumors show an organoid pattern with high vascularity, but usually also show other features like a trabecular pattern, pseudoglandular acini, and stippled nuclei. Keratin immunostain is positive in the majority of carcinoid tumors, but negative in paragangliomas. Positivity for neuroendocrine markers on immunohistochemistry, and neurosecretory granules on electron microscopy, are not helpful since paraganglioma shares these features with carcinoid tumor. Glomus tumor is another tumor to distinguish from paraganglioma, again because of its nesting pattern and vascularity. The differential point lies mainly on smooth muscle features in glomus cells. The prognosis for tracheal paragangliomas has been reported to be good, with no case of recurrence reported after surgical excision. Two cases of thyroid paraganglioma have been reported as behaving in an invasive manner, with secondary involvement of the trachea.42,63 A paraganglioma arising in the superior mediastinum between the trachea and left subclavian artery, displacing these structures and extending upward to the neck, has also been reported.62
Glomus Tumor This benign neoplasm, which primarily affects the skin, is rare in the trachea. There are approximately 14 cases reported in the literature, and we have encountered a case at Massachusetts General Hospital. The patients were in their fourth to seventh decades of life, and most were men. The tumor usually produces a polypoid mass, protruding into the lumen, and causes partial obstruction. Histologically, and similar to other sites, the tumor shows nests of round monotonous cells with bland central nuclei and amphophilic or lightly eosinophilic cytoplasms, separated by ectatic thin-walled vascular channels (hence the synonymous term “glomangioma”) (Figures 3-55, 3-56 [Color Plate 7]). The tumor cells, in some cases, form solid sheets with inconspicuous vascular channels and little stroma. The stroma is sometimes prominent with hyaline character, in which the tumor cells are embedded. One case of an oncocytic glomus tumor showing intensely eosinophilic cytoplasm has been reported.64 The glomus cells, which are believed to be modified smooth muscle cells of the glomus body, are positive for desmin and smooth muscle actin immunohistochemically. Ultrastructural features include numerous pinocytotic vesicles, intracytoplasmic bundles of myofibrils, and focal electron dense bodies.65
Pathology of Tracheal Tumors
The major differential diagnosis is carcinoid tumor, to which the cytology and architecture may have close resemblance.66 The distinguishing features are the presence of neuroendocrine differentiation in carcinoid tumor, and the absence of smooth muscle differentiation on immunohistochemical and electron microscopic examinations. The behavior is benign, with no recurrence reported in the literature. The glomus tumor that we saw recurred 3 years after initial surgery.
Lymphomas Lymphomas are exceedingly rare as primary tumors of the trachea. Somewhat more common is secondary involvement by a nodal lymphoma.
Non-Hodgkin’s Lymphoma Most of the cases of non-Hodgkin’s lymphoma are secondary involvement by a nodal lymphoma. In the majority of these cases, neoplastic lymphoid tissue from peritracheal lymph nodes infiltrates the tracheal wall, causing stenosis of the tracheal lumen. Less frequently, non-Hodgkin’s lymphoma may arise in the trachea. Excluding plasmacytomas, only about 10 cases of primary tracheal non-Hodgkin’s lymphoma have been reported.67 A few cases of direct invasion from adjacent structures such as the thyroid or thymus are also reported.68 Biology. Both primary and secondary tracheobronchial lymphomas occur in both genders, generally in the sixth and seventh decades.67,69 Pathology. Grossly, tumoral tissue may infiltrate the wall, causing narrowing and stenosis of the lumen, or protrude into the lumen as a polypoid mass.67,69 A hom*ogeneous, pale-tan, and fleshy surface is seen on cut section (Figure 3-57 [Color Plate 8]). Microscopic appearance shows sheets of round or oval lymphoid cells, the details of which depend on the lymphoma type. In both secondary and primary lymphomas, low-grade lymphomas outnumber others. In primary lymphomas, low-grade B-cell lymphoma of mucosa associated lymphoid tissue (MALT), small lymphocytic lymphoma, lymphoplasmacytic lymphoma, and plasmacytoma have been reported. The changes in classification of non-Hodgkin’s lymphomas, and increasing diagnostic accuracy with the advent of immunohistochemical and molecular genetic techniques, have affected the diagnosis and categorization of lymphoma cases. Some of these cases may have been categorized under “undifferentiated carcinoma” or “pseudolymphoma” in the past. Major differential diagnoses include Hodgkin’s lymphoma, small cell carcinoma, undifferentiated carcinoma, and benign exuberant lymphoid proliferation (the so-called “pseudolymphoma”). The frequency of progression of primary tracheal lymphoma to systemic lymphoma is not established. In the absence of systemic lymphoma, cases can be treated by surgery or by radiation.
Hodgkin’s Lymphoma Hodgkin’s lymphomas occur in the trachea (Figure 3-58 [Color Plate 8]) mostly through direct extension of adjacent peritracheal lymph nodes, involved by the tumor, either at first presentation or in recurrence.70,71 Many of the reported cases are in children. Tracheal involvement by a thymic Hodgkin’s lymphoma has also been reported.72
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Farrell ML, Michael L. Tracheal chondrosarcoma: a case report. Head Neck 1998;20:568–72. Dennie CJ, Coblentz CL. The trachea: pathologic conditions and trauma. Can Assoc Radiol J 1993;44:157–67. Bailey CM, Froehlich P, Hoeve HL. Management of subglottic hemangioma. Laryngol Otol 1998;112:765–8. Messineo A, Wesson DE, Filler RM, Smith CR. Juvenile hemangiomas involving the thoracic trachea in children: report of two cases. J Pediatr Surg 1992;27:1291–3. Schwartz MZ. Maintenance of the placental circulation to evaluate and treat an infant with massive head and neck hemangioma. J Pediatr Surg 1993;28:520–2. Yuasa K, Shimizu T, Toyoda T. The giant cavernous hemangioma of the neck. Nippon Kyobu Geka Gakkai Zasshi 1992;40:1274–8. Mills SE, Cooper PH, Fechner RE. Lobular capillary hemangioma: the underlying lesion of pyogenic granuloma. A study of 73 cases from the oral and nasal mucous membranes. Am J Surg Pathol 1980; 4:470–9. Belda J, Canalis E, Gimferrer JM, et al. Subglottic stenosis in an HIV positive patient: an exceptional form of clinical presentation in Kaposi’s sarcoma. Eur J Cardiovasc Surg 1997;11:191–3. Miller RF, Tomlinson MC, Cottoril CP, et al. Bronchopulmonary Kaposi sarcoma in patients with AIDS. Thorax 1992;47:721–5. Sleiman C, Mal H, Roue C, et al. Bronchial Kaposi’s sarcoma after single lung transplantation. Eur Resp J 1997;10:1181–3. Rajaratnam K, Desai S. Kaposi’s sarcoma of the trachea. J Laryngol Otol 1988;102:951–3. Gavilan J, Rodriguez-Peralto JL, Tomas MD, et al. Hemangiopericytoma of the trachea. J Laryngol Otol 1987;101:738–42. Tytor T, Olofsson J. Thyroid tumors invading the larynx and trachea. J Otolaryngol 1986;15:74–9. f*ckai I, Masaoka A, Yamakawa Y, et al. Mediastinal malignant epithelial schwannoma. Chest 1995;108:574–5. Meredith HC, Valicenti JF. Solitary neurofibroma of the trachea. Br J Radiol 1978;51:218–9. Lossos IS, Breuer R, Lafair JS. Endotracheal neurofibroma in a patient with von Recklinghausen’s disease. Eur Respir J 1988;1:464–5. Fischberg C, Cotting J, Hack I, et al. Fatal double tracheoesophageal vascular compression and neurofibromatosis. Arch Pediatr 1996;3:1253–7. Horovitz AG, Khalil KJ, Verani RR, et al. Primary intratracheal neurilemoma. J Thorac Cardiovasc Surg 1983;85:313–7. Rusch VW, Schmidt RA. Tracheal schwannoma: management by endoscopic laser resection. Thorax 1994; 49:85–6. Yano T, Hara N, Ichinose Y, et al. An intrathoracic vagus nerve schwannoma invading the trachea. Surg Today 1993;23:1113–5. Burton DM, Heffner DK, Patow CA. Granular cell tumor of the trachea. Laryngoscope 1992;102:807–13. Desai RC, Holinger LD, Gonzalez-Crussi F. Tracheal neoplasms in children. Ann Otol Rhinol Laryngol 1998;107:790–6. Gorman RC, Lights V, Furth E, Torosian MH. Unique presentation of granular cell tumor as a paratracheal mass. Oncol Rep 1998;5:1551–4. Kintanar EB, Giordano TJ, Thompson NW, Michael CW. Granular cell tumor of the trachea masquerading as Hurthle-cell neoplasm on fine-needle aspirate: a case report. Diagn Cytopathol 2000;22:379–89. Yang SW, Hong SW, Cho MW, Kang SJ. Malignant granular cell tumor at the retrotracheal space. Yonsei Med J 1999;40:76–9.
Pathology of Tracheal Tumors
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Amble FR, Olsen KD, Nascimento AG, Foote RL. Head and neck synovial cell sarcoma. Otolaryngol Head Neck Surg 1992;107:631–7. Sykes At, Rokkas CK, Kajdacsy-Balla A, Haasler GB. Primary tracheal synovial cell sarcoma: a first case report. J Thorac Cardiovasc Surg 1997;114:678–80. Nakayama DK, Harrison MR, de Lorimier AA, et al. Reconstructive surgery for obstructive lesions of the intrathoracic trachea in infants and small children. J Pediatr Surg 1982;17:854–68. Gross E, Chen MK, Hollabagh RS, Joyner RE. Tracheal hamartoma: report of a child with a neck mass. J Pediatr Surg 1996;31:1584–5. Suzuki N, Ohno S, Ishii Y, Kitamura S. Peripheral intrapulmonary hamartoma accompanied by a similar endotracheal lesion. Chest 1994;106:1291–3. Dominguez H, Hariri J, Pless S. Multiple pulmonary chondrohamartomas in trachea, bronchi and lung parenchyma. Respir Med 1996;90:111–4. Sing TM, Wong KP, Young N, Despas P. Chemodectoma of the trachea. Thorax 1996;51:341–2. Gallimore AP, Goldstraw P. Tracheal paraganglioma. Thorax 1993;48:866–7. Mitsudo SM, Grajower MM, Balbi H, Silver C. Malignant paraganglioma of the thyroid gland. Arch Pathol Lab Med 1987;111:378–80. Shin DH, Park SS, Lee LH, et al. Oncocytic glomus tumor of the trachea. Chest 1990;98:1021–3.
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Ito H, Motohiro K, Nomura S, Tahara E. Glomus tumor of the trachea. Immunohistochemical and electron microscopic studies. Pathol Res Pract 1988;183:778–84. Menaissy YM, Gall AA, Mansour KA. Glomus tumor of the trachea. Ann Thorac Surg 2000;70:295–7. Fidias P, Wright C, Harris NL, et al. Primary tracheal nonHodgkin’s lymphoma. Cancer 1996;77:2332–8. Zahradka W, Wuttke WD, Gehrandt F. Primary lymphoblastic lymphoma of the thyroid. Z Gesamte Inn Med 1990;45:58–60. Kaplan MA, Pettit CL, Zukerberg LR, Harris NL. Primary lymphoma of the trachea with morphologic and immunophenotypic characteristics of low-grade B-cell lymphoma of mucosa-associated lymphoid tissue. Am J Surg Pathol 1992;16:71–5. Case records of the Massachusetts General Hospital. Weekly clinicopathological exercises. Case 43-1984. A 35-year-old woman with tracheal stenosis after treatment for Hodgkin’s disease. N Engl J Med 1984; 311:1105–13. Tanka H, Nakahara K, Sakai S, et al. A case of Hodgkin’s disease with endotracheal tumor presenting with severe airflow obstruction. Nippon Kyobu Shikkan Gakkai Zasshi 1992;30:1732–7. Fujimura Y, Kobayashi T, Nawata S, et al. A surgical case of Hodgkin’s lymphoma originated from thymus. Nippon Kyobu Geka Gakkai Zasshi 1990;38:126–9.
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3C TUMOR-LIKE LESIONS OF THE TRACHEA Eugene J. Mark, MD Javad Beheshti, MD
Inflammatory Myofibroblastic Tumor (Inflammatory Pseudotumor) Intratracheal Thyroid Amyloidosis Rheumatoid Nodule Tracheopathia Osteoplastica
There are certain infectious, inflammatory, or reactive processes that can cause various degrees of tracheal lumen obstruction by focal nodular or polypoid protrusion into the lumen, or more extensive tracheal wall thickening and luminal narrowing. Infections such as tuberculosis and histoplasmosis, inflammatory lesions such as sarcoidosis, and reactive processes such as post-traumatic granulation tissue formation and fibrosis are amongst these conditions. Herein, we address several of these lesions that merit special consideration on histopathological grounds, and because they are more likely to be mistaken for tracheal tumors on endoscopy.
Inflammatory Myofibroblastic Tumor (Inflammatory Pseudotumor) Inflammatory myofibroblastic tumor, also called “inflammatory pseudotumor,” “plasma cell granuloma,” and a battery of other names, is a pseudosarcomatous proliferation of myofibroblastic cells forming a mass lesion. The most common site is the lung, but cases in many other organs including skin, soft tissues, and gastrointestinal tract have been reported. Various etiologies such as inflammatory stimuli and trauma have been proposed, but current data suggest a benign neoplastic process in the lung and in many other organs.1,2 This differs from myofibroblastic proliferation of the genitourinary tract, which is believed to be reactive and non-neoplastic.3 Although these lesions in the trachea may be neoplastic, like pulmonary inflammatory myofibroblastic tumors, the exact nature remains to be clarified. Biology. Inflammatory pseudotumor of the trachea has been reported in all ages, from infancy and childhood to late adulthood.4–7 It affects both men and women. Pathology. Inflammatory myofibroblastic tumor typically produces a rather circ*mscribed polypoid mass in the trachea, which can be sessile or pedunculated.3
Pathology of Tracheal Tumors
Microscopically, the lesion is located in the tracheal wall and is covered by reactive or ulcerated epithelium. It consists of proliferated spindle cells arranged more or less in fascicular pattern. These cells contain regular nuclei with open chromatin and minimal pleomorphism. Mitotic figures are not abundant. A variety of inflammatory cells, including lymphocytes, plasma cells, histiocytes, neutrophils, and eosinophils, are infiltrated. Foci of myxoid change may be present. Necrosis does not occur. Differential diagnosis includes a number of benign and malignant soft tissue tumors such as fibromatosis, benign fibrous histiocytoma, fibrosarcoma, leiomyoma, leiomyosarcoma, neurogenic tumors, hemangiopericytoma, or less frequently, other spindle cell tumors. As the designation “pseudosarcomatous” implies, the histology may closely resemble spindle cell sarcomas. The weak or focally positive immunohistochemical staining for desmin and smooth muscle actin, which is due to myofibroblastic differentiation, distinguishes this lesion from fibromatosis, fibrous histiocytoma, fibrosarcoma, and hemangiopericytoma, all of which lack these markers. The positivity for desmin and smooth muscle actin is much stronger in smooth muscle tumors. The negative immunostain for S-100 antigen helps in differentiation from neurogenic tumors, which are positive for this marker. Although malignant fibrous histiocytoma is generally among the differential diagnoses, the absence of marked pleomorphism, high mitotic activity, and necrosis rules out this tumor and other high-grade sarcomas.
Intratracheal Thyroid Although total thyroid ectopia is an extremely rare anomaly, ectopic thyroid tissue has been reported in different organs with the normal thyroid gland in place as well. These include lingual, sublingual, thyroglossal, laryngeal, tracheal, mediastinal, and diaphragmatic sites. Although more than 100 cases were published in Europe, mostly from Germany, only about 25 cases have been reported in the Englishlanguage literature.8 Biology. In a review of 23 cases, the patients ranged in age from newborn to 56 years, with a mean of 28.3 years, but most were young adults.9 Females outnumbered males in a ratio of 3.8:1. Clinical presentation varied from asymptomatic cases found on autopsy, to symptoms such as cough, wheezing, stridor, and dyspnea. Symptoms worsened in 3 patients during pregnancy, 1 of them also with menses.9,10 Another patient had increasing wheezing during labor and died postpartum because she had been treated for asthma and was not suspected to have an intratracheal mass.10 Increased dyspnea was noted in a girl with menarche. These have led some to suggest hormonal stimulation as a basis for ectopic tissue enlargement during phases of altered female sex hormone production. Several theories have been proposed to explain aberrant location of thyroid tissue including “malformation” and “ingrowth ” theories.11 Pathology. Grossly, most of the cases are single or multiple nodules or plaques in the tracheal wall, often visible endoscopically as submucosal masses. These usually measure 1 to 3 cm in dimension but may be as large as 5 cm.12 The most common location is the posterolateral wall of the upper trachea. Microscopically, this condition often consists of normal thyroid tissue with follicles and intervening stroma. Goiterous enlargement has been observed in some cases, with an associated thyroid gland goiter in 75% of cases.13 Connection to the thyroid gland with a strand of thyroid tissue has been seen in some cases. Malignant change may occur in the intratracheal thyroid, and poses a diagnostic problem to the pathologist as to whether the lesion has arisen from intratracheal thyroid or represents invasion by thyroid gland carcinoma. Much more common is direct invasion of papillary carcinoma of the thyroid, usually through the tracheal anterolateral wall directly into the tracheal lumen, where it may cause obstruction.
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The area of penetration is between the cartilaginous plates, where blood vessels, lymphatics, and nerves course perpendicular to the tracheal lumen.14 Biopsy of such tumors may show the characteristic features of papillary carcinoma of the thyroid, for example, nuclear grooves, nuclear overlap with molding, and intranuclear pseudoinclusions. Because of the possibility of surgical resection of a papillary carcinoma invading the trachea in the absence of distant metastases, a staging system has been devised (see Chapter 8, “Secondary Tracheal Neoplasms”).14
Amyloidosis Amyloidosis may involve any site in the upper or lower respiratory tract, the larynx being the most common site. It may also affect the esophagus and mediastinal nodes, or produce mass lesions in the neck. Tracheal involvement may occur as part of a generalized amyloidosis, whether primary or secondary, or in a localized form, which itself can be primary or secondary to plasmacytoma. Biology. The primary localized form affects adults, and only exceptional cases are reported in children.15 The other forms follow the age distribution of generalized amyloidosis and solitary tracheal plasmacytomas. Tracheal obstruction may occur in severe forms and the obstruction may lead to infections and respiratory failure. Bleeding, although not frequent, may be massive and even fatal, as occurred in a patient with laryngeal amyloidosis.16 The bleeding problem is probably related to blood vessels, which themselves may have amyloid in their walls and may not constrict normally. Pathology. Gross appearance, as seen on bronchoscopy, varies from diffusely heaped up edematous mucosa to single or multiple nodules protruding into the lumen. Macroscopically, tracheal amyloidosis has a firm grey, waxy cut surface (Figure 3-59 [Color Plate 8]). Microscopically, nodules of amorphous, lightly eosinophilic material are deposited in the mucosa (Figure 3-60 [Color Plate 8]) and large deposits may erode into cartilage. Scant chronic inflammation and fibrosis may be present. Vascular amyloidosis may be noted. Where amyloid extends deeply, it may involve the mucosal glands, producing globules of eosinophilic material replacing the atrophic gland acini. Cartilaginous change, calcification, and osseous metaplasia may occur. Foreign body giant cells and granulomas often surround the amyloid. In localized tracheal amyloidosis secondary to solitary plasmacytoma, amyloid material usually comprises a minority of the tumoral tissue, but it may be even more extensive than the neoplastic plasma cells. Histochemical staining with Congo red shows apple-green birefringence under polarized light, which confirms the diagnosis (Figure 3-61 [Color Plate 8]). Other stains such as methyl violet or thioflavin-T may also be used. Electron microscopy shows the typical fibrillar structure of amyloid. Immunoglobulin light chains were present and mixed with amyloid in one case. Bronchoscopic differential diagnoses include tracheopathia osteoplastica, relapsing polychondritis, inflammatory lesions, and tumors. With regard to histology, amyloid deposits may be replaced with cartilage and bone. It must be distinguished from other lesions with cartilage and bone, particularly tracheopathia osteoplastica, but also from degenerative and reparative changes that can occur with tracheomalacia. Foreign body reaction to the amyloid should not be mistaken for granulomatous diseases like tuberculosis and sarcoidosis. Prognosis is generally good in primary localized forms, but the risk of bleeding exists. Laser treatment or surgical excision may be necessary in obstructive lesions. Nodular tracheal amyloidosis may cause obstruction, and postobstructive infection may develop. In diffuse tracheobronchial amyloidosis, postobstructive infection and respiratory failure may occur. In generalized amyloidosis, pulmonary involvement with pulmonary hypertension, cardiac involvement with arrhythmias and failure, or other complications may complicate the clinical course.
Pathology of Tracheal Tumors
Rheumatoid Nodule A case of tracheal rheumatoid nodules in a 45-year-old Chinese man has been reported.17 The patient had suffered rheumatoid arthritis for 7 years, with articular manifestations and deformities. Subcutaneous rheumatoid nodules had been present over his elbows and hands. Endoscopy showed four smooth, whitish nodules, each 3 to 5 mm in diameter. Further specification was not possible on endoscopic findings. Histologic examination showed foci of necrobiosis and vascular fibrinoid necrosis in the vessels surrounded by palisading histiocytes. Among the histologic differential diagnoses are necrotizing granulomatous diseases such as tuberculosis and histoplasmosis. Amyloid deposits must also be separated from central necrobiosis of this lesion, which may have a similar eosinophilic patternless aspect. Amyloid material, however, is more hom*ogeneous, and stains with Congo red and other special histochemical stains.
Tracheopathia Osteoplastica Tracheopathia osteoplastica, also known as “tracheobronchopathia osteochondroplastica,” is an infrequent disease of the trachea and major bronchi. It produces multiple osseous and cartilaginous projections into the airway lumen. Biology. The disease affects an age range from 12 to 72 years, with the peak being in the fifth and sixth decades. No gender predominance has been present. Familial occurrence is exceptional but is reported in a mother and daughter.18 Various theories have been proposed to explain the pathogenesis of this disease, including the outgrowth of normal cartilaginous rings causing ecchondrosis and exostosis, and abnormalities in tracheal elastic tissue causing metaplastic cartilage and bone formation.19,20 More recently, chronic infection, chemical or mechanical irritation, metabolic abnormalities, and genetic predisposition have been postulated, but the exact cause is unknown.18,21 Although some earlier reports have associated this disease with tracheobronchial amyloidosis, most patients lack amyloid deposits in airways.22,23 Many patients are asymptomatic, especially early in the course and in localized forms. Symptomatic patients may have dyspnea, cough, wheezing, stridor, or hemoptysis. Postobstructive pneumonia may develop if significantly symptomatic cases are not diagnosed early in their course.18 Pathology. Grossly, lesions are seen bronchoscopically as irregular whitish nodules carpeting the airway lumen, producing a “rock garden” appearance (Figure 3-62 [Color Plate 8]). It is most prominent in the lower two-thirds of the anterolateral tracheal wall and often extends to major bronchi. Microscopically, cartilaginous and osseous submucosal nodules are seen, covered by a normal or metaplastic squamous epithelium (Figures 3-63, 3-64 [Color Plate 8]). Foci of calcification are sometimes seen. Inflammation is usually absent or sparse, and surface ulceration is not extensive. Differential diagnoses of bronchoscopic findings include papillomatosis and tumors (especially in localized forms of tracheopathia osteoplastica), and infections such as tuberculosis, sarcoidosis, and others. Microscopic differential diagnosis may include relapsing polychondritis. Osseous metaplasia has also been seen in carcinomas, carcinoid tumors, and tuberculosis. Multinodular amyloidosis may simulate tracheopathia osteoplastica when it has secondary ossification, or it may be part of tracheopathia osteoplastica. The lack or scarcity of inflammation in tracheopathia osteoplastica, and the continuity of a strutwork of osteocartilaginous nodules inner to the tracheal cartilages, help in its differentiation from inflammatory lesions and post-traumatic ossification. Prognosis is usually good, particularly in asymptomatic and localized lesions. Tracheoplasty or surgical resection may be required in extensive and obstructive lesions (see Chapter 14, “Infectious, Inflammatory, Infiltrative, Idiopathic, and Miscellaneous Tracheal Lesions” and Chapter 32, “Surgery for Tracheomalacia, Tracheopathia Osteopathica, Tracheal Compression, and Staged Reconstruction of the Trachea”).
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References 1.
2. 3. 4. 5. 6. 7. 8. 9. 10. 11.
Pettinato G, Manivel JC, De Rosa N, Dehner LP. Inflammatory myofibroblastic tumor (plasma cell granuloma). Clinicopathologic study of 20 cases with immunohistochemical and ultrastructural observations. Am J Clin Pathol 1990;94:538–46. Weiss SW, Goldblum JR, editors. Inflammatory myofibroblastic tumor. In: Enzinger and Weiss’s soft tissue tumors. St Louis: Mosby; 2001. p. 383–9. Weiss SW, Goldblum JR, editors. Benign fibrous tissue tumors. In: Enzinger and Weiss’s soft tissue tumors. St. Louis: Mosby; 2001. p. 274–5. Dewar AL, Connett GJ. Inflammatory pseudotumor of the trachea in a ten-month-old infant. Pediatr Pulmonol 1997;23:307–9. Aijaz F, Salam AU, Muzaffar S, et al. Inflammatory pseudotumor of the trachea: report of a case in an eight year old child. J Laryngol Otol 1994;108:613–6. Barker AP, Carter MJ, Matz LR, Armstrong JA. Plasma cell granuloma of the trachea. Med J Aust 1987;146:443–5. Ishii Y, Inoue F, Kamikawa Y, et al. A case report of tracheal inflammatory pseudotumor. Nippon Kyobu Geka Gakkai Zasshi 1993;41:672–7. Myers EN, Panlango IP Jr. Intratracheal thyroid. Laryngoscope 1975;85:1833–40. Brandwein M, Som P, Urken M. Benign intratracheal thyroid, a possible cause for preoperative overstaging. Arch Otolaryngol Head Neck Surg 1998;124:1266–9. Ferlito A, Giarelli L, Silvestri F. Intratracheal thyroid. J Laryngol Otol 1988;102:95–6. Chanin LR, Greenberg LM. Pediatric upper airway obstruction due to ectopic thyroid: classification and case reports. Laryngoscope 1998;98:422–7.
12. 13. 14.
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Thoren L. Intratracheal goiter. Acta Chir Scand 1947; 95:455–512. Dowling EA, Johnson IM, Collier FD, et al. Intratracheal goiter: a clinicopathological review. Ann Surg 1962;156:258–67. Shin DH, Mark EJ, Suen MC, Grillo HC. Pathologic staging of papillary carcinoma of the thyroid with airway invasion based on the anatomic manner of extension to the trachea. Hum Pathol 1993;24:866–70. Plaza Martos JA, Garcia MH, Garzon MA. Amyloid tumor of the trachea. Presentation of a pediatric case. Ann Esp Pediatr 1989;30:61–2. Chow LT, Chow WH, Shum BS. Fatal massive upper respiratory tract hemorrhage: an unusual complication of localized amyloidosis of the larynx. J Laryngol Otol 1993;107:51–3. Ip SM, Wong MP, Wong KL. Rheumatoid nodules in the trachea. Chest 1993;1:301–3. Young RH, Sandstrom RE, Mark GJ. Tracheopathia osteoplastica. J Thorac Cardiovasc Surg 1980;79:537–41. Virchow R. Die Krankhaften Gesch wulste. Berlin: Hirschwald 1863;1:442–3. Aschoff-Freiburg L. Ueber tracheopathia osteoplastica. Ver Dtsch Ges Pathol 1910;14:125–7. Prakash UBS, McCullogh AE, Edell ES, Nienhuis DM. Tracheobronchopathia osteochondroplastica: familial occurrence. Mayo Clin Proc 1989;64:1091–6. Sakula A. Tracheobronchopathia osteochondroplastica: its relationship to primary tracheobronchial amyloidosis. Thorax 1968;23:105–10. Martin CJ. Tracheobronchopathia osteochondroplastica. Arch Otolaryngol 1974;100:290–3.
CHAPTER FOUR
Imaging the Larynx and Trachea Jo-Anne O. Shepard, MD Alfred L. Weber, MD
Normal Anatomy Imaging Technique Congenital Abnormalities Postpneumonectomy Syndrome Trauma Saber-Sheath Trachea Granulomatous Lesions
Other Benign Infiltrative Lesions Benign Tumors and Cysts Malignant Lesions Tracheobronchomegaly Tracheobronchomalacia Acquired Tracheobronchoesophageal Fistulae
Normal Anatomy Larynx The larynx is divided into supraglottic, glottic, and subglottic parts, whereas the trachea is composed of the cervical extrathoracic trachea and the mid and lower intrathoracic trachea.1,2 The supraglottic portion of the larynx is constituted by the epiglottis, aryepiglottic folds, arytenoids, and false cords. The glottic portion of the larynx is made up of the laryngeal ventricles and both vocal cords. The crescent-shaped laryngeal ventricles are situated between the false and true cords as lateral invagin*tions of mucosa. The subglottic space extends from the undersurface of the vocal cords to the inferior margin of the cricoid cartilage, which is also the lower boundary of the larynx. This subglottic area is oval in shape and measures about 1.5 to 2 cm in length (Figures 4-1 through 4-4).3–5
Trachea The cervical trachea extends from the inferior margin of the cricoid cartilage to the thoracic inlet, and the intrathoracic trachea extends from the thoracic inlet to the carina where it divides into the right and left main bronchi.6,7 There are 14 to 22 (mean 17) C-shaped hyaline tracheal cartilage rings that support the anterior and lateral tracheal walls. The cartilaginous portion of each tracheal ring forms a “C” with the membranous portion found posteriorly, which is unsupported by cartilage. The first ring is partly recessed into the broader ring of the cricoid cartilage. The cartilaginous rings are usually semicircular or horseshoeshaped and are the chief determinant of cross-sectional shape. Tracheal diameter grows from 3 to 4 mm in infancy to about 20 mm in adulthood.8,9 The cervical trachea is subject to atmospheric pressure and the intrathoracic trachea is subject to intrathoracic pressure that is equivalent to pleural pressure during quiet breathing. During forced expiration or coughing, the intrathoracic pressure becomes greater than atmospheric pressure, increasing the compressive transmural pressure that narrows the intrathoracic trachea.
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4-1 Normal laryngeal anatomy. Normal lateral view of the neck demonstrates the epiglottis, aryepiglottic folds (AEF), false vocal cords (FC), true cords (VC), and subglottic space (asterisk). FIGURE
FIGURE 4-2 Normal laryngeal anatomy. Anteroposterior high-kilovoltage view of the larynx and trachea demonstrates the aryepiglottic folds (AEF), false cords (FC), vocal cords (VC), and subglottic space (lower asterisk). Upper asterisk = laryngeal ventricle; plus sign = pyriform sinus.
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FIGURE 4-3
Normal laryngeal anatomy. A, Axial computed tomography (CT) scan, at the upper third of larynx level, outlines the epiglottis and aryepiglottic folds (AEF). B, Axial CT scan, at the vocal cord (VC) level, defines the arytenoids, cricoid cartilage, and thyroid cartilage.
A
A
B
B
4-4 Normal laryngeal anatomy. A, Normal lateral T1-weighted image of the larynx and cervical trachea illustrates the epiglottis, preepiglottic fat space (asterisk), and subglottic space (plus sign). B, Coronal T1-weighted image of the larynx and cervical trachea depicts the vocal cord (VC) and subglottic space (asterisk).
FIGURE
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Coronal narrowing occurs due to inward bending of the cartilaginous rings, and sagittal narrowing occurs as a result of invagin*tion of the posterior membranous wall. Narrowing of the tracheal diameter up to 50% may be considered normal. Accentuated tracheal collapse may occur if there is abnormal flaccidity as occurs in tracheomalacia. Normal reflections of the trachea can be identified on chest radiographs.10,11 The right paratracheal stripe (RPS) is a thin stripe formed by the reflection of the right upper lobe with the right tracheal wall, normally 1 to 4 mm in thickness. Widening of the RPS is indicative of inherent tracheal wall disease or widening of the paratracheal soft tissues or right mediastinal pleural reflection. Normally, the left tracheal wall is not discernible because it has no contact with the left lung. The tracheoesophageal stripe (TES) is formed by the posterior tracheal wall, the anterior wall of the esophagus, and interposed fat and connective tissues, and is seen only when the esophagus contains air. The posterior tracheal band (PTB) is comprised of the posterior membranous wall of the trachea, which is formed by the interface of air in the tracheal lumen and the aerated retrotracheal recess of the right upper lobe. It is present only from the thoracic inlet to the carina, whereas the TES can be discerned from the cervical and intrathoracic regions. The PTB and TES are seen on the lateral chest radiographs, in 80 to 90% of patients. They have a uniform width of 3 to 5 mm. A left-sided aortic arch will make an impression on the left lower tracheal wall. A right-sided aortic arch will make a similar impression on the right lower tracheal wall. The tracheal bifurcation is at the level of the fifth thoracic vertebra. The intrathoracic esophagus lies slightly to the left and behind the trachea. The azygous vein may be seen as a horizontal soft tissue band, which crosses posterior to the trachea toward the right tracheobronchial angle.
Imaging Technique Radiography Anteroposterior and Lateral Films of the Neck Including Cervical Trachea. Routine radiologic investigation of the larynx and cervical trachea is composed of anteroposterior (AP) and lateral films of the neck (see Figures 4-1, 4-2) and oblique views of the trachea with the patient in a 45° to 60° rotation.12,13 The lateral view of the neck is obtained with the head slightly hyperextended to bring the larynx and upper trachea up from the retrosternal position. This lateral view provides useful information about the base of tongue, vallecular area, thyroid and cricoid cartilages, intralaryngeal structures (including the epiglottis, aryepiglottic folds, arytenoids, false cords, ventricles, true cords, and subglottic space), posterior pharyngeal wall, and precervical soft tissues. Diseases that arise or spread in the sagittal plane, including the anterior and posterior tracheal wall, are readily visible. The frontal AP view of the larynx and trachea is obtained by using a highkilovoltage technique (120 kV) and placing a 1 mm copper filter in front of the x-ray tube.14 This view provides a survey of the entire airway from the hyoid bone to the tracheal bifurcation and main bronchi. This technique enhances the air–soft tissue interface by obscuring bone shadows. The frontal projection aids in lateralizing disease processes and supplements the lateral view. In cases of a suspected foreign body, a lateral view during swallowing is added to distinguish a foreign body from the calcified cartilaginous structures of the larynx, which move upward. This swallowing film also allows visualization of a foreign body in the upper esophagus that has been obscured by the soft tissue structures of the superimposed shoulders. One to 2 cm of the trachea and esophagus ascend out of the mediastinum during swallowing, depicting additional trachea obscured by soft tissue of the thoracic inlet. As much as 2 to 3 cm of the trachea can move above the suprasternal notch in hyperextension of the neck. This is age dependent and decreases progressively in older patients, especially if they suffer from chronic obstructive pulmonary disease. Radiography of the Intrathoracic Trachea. The chest radiograph is the traditional screening study of the trachea. Posteroanterior (PA) and lateral views of the chest are routinely employed. The distal cervical and
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intrathoracic portions of the trachea are visible on both views; however, overlying mediastinal and bony structures often obscure intrathoracic tracheal abnormalities. Bilateral oblique chest radiographs rotate the spine and mediastinal structures so that the trachea and carina are less obscured (Figure 4-5). Highkilovoltage radiographic technique can improve the visualization of intrinsic airway lesions.6 Digital radiography can improve the visibility of tracheal walls and mediastinal reflections by virtue of edge enhancement techniques.7 Conventional tomography of the trachea is no longer routinely employed, having been replaced by multidetector computed tomography (CT) scanning.15
Fluoroscopy Fluoroscopy of the Larynx and Cervical Trachea. In order to assess the dynamics of the larynx and cervical trachea, fluoroscopy in the sitting position is indicated.12 It supplements all other radiologic studies of the larynx and trachea, including CT scans. A thorough knowledge of the normal roentgenographic anatomy of the larynx and of functional changes encountered during different phonation maneuvers is a prerequisite. Assessment of vocal cord motion is important in the staging of malignant tumors of the larynx. Fixation of the vocal cords or paralysis of the cords from other causes (eg, thyroid carcinoma, lung cancer with mediastinal extension, aortic aneurysm, or iatrogenic trauma) can be assessed easily with phonation maneuvers such as “E” and inspiration. Opening of the ventricles can be accomplished by having the patient phonate “E” during inspiration (reversed “E”). The aryepiglottic folds, true and false cords, and ventricles are always symmetrical. The subglottic space is tubular in shape and is limited superiorly by the vocal cords, which form a right angle with the lateral wall of the subglottic space. The lumen of the subglottic
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FIGURE 4-5 Tracheal adenoid cystic carcinoma. Posteroanterior (A) and oblique (B) views of the chest. A focal soft tissue mass is evident in the midtrachea, superimposed on the thoracic spine on the posteroanterior view and better visualized on the oblique view.
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space is well marginated, and is oval to round in shape. Asymmetry of the pyriform sinuses is a common finding; however, the medial walls of the pyriform sinuses are usually symmetrical. In the assessment of lesions of the larynx and cervical trachea, the following radiologic parameters should be determined: 1) the location, extent, size, and density of the lesion; 2) definition of the margin, presence of calcification, degree of airway compromise, cartilaginous abnormalities including destruction, and invasion of contiguous structures; 3) distensibility of the pyriform sinuses and ventricles; 4) mobility of the true and false cords; 5) displacement or tilt of the larynx; 6) extralaryngeal masses; 7) calcifications; and 8) presence of an air-filled sac. In the fluoroscopic study of the larynx, the air–soft tissue interface can be increased easily by mounting a 1 mm copper filter on the tabletop in the field of the x-ray beam. This is especially useful in studying the infant larynx in the frontal view, for assessment of the vocal cords and subglottic space with suspected pathology in this region (eg, hemangioma, cyst, or subglottic stenosis). Simultaneously, spot films on the cervical trachea with different degrees of rotation are taken to free the trachea from superimposed normal anatomic structures at the thoracic inlet. Fluoroscopy of the Trachea. The dynamic changes of the tracheal wall cannot be evaluated on static imaging.16 The nature and severity of tracheal caliber changes are best observed during real-time imaging, such as fluoroscopy. Tracheal compliance can be evaluated by the Valsalva, modified Valsalva, and Müller maneuvers and by coughing. During the Valsalva maneuver, the patient takes a deep inspiration and performs forced expiration against a closed glottis. The modified Valsalva maneuver simulates the action of blowing up a balloon. With both of these maneuvers, a weakness of the cervical trachea will manifest as a bulge (eg, laryngocele or pharyngocele). The Müller maneuver is a sniff test that consists of a forceful inspiration through the nose. With this maneuver, the pleural pressure becomes more negative than with normal inspirations, causing the intrathoracic trachea to widen more than with normal inspiration and the cervical trachea to collapse to a greater degree. When there is obstruction of the cervical trachea, inspiratory collapse may occur during forced inspiration because the pressure around the cervical trachea exceeds the intratracheal pressure. The exact opposite will occur in the intrathoracic trachea. During coughing or forced expiration, the pressure outside the airways is greater than the pressure inside, resulting in a compressive force to the intrathoracic trachea. As a result, expiratory collapse is commonly found in tracheobronchomalacia and in peripheral obstructive airway diseases such as asthma, bronchitis, or bronchiolitis (Figure 4-6).
Barium Esophagogram The barium esophagogram is an important component of the work-up of congenital and acquired lesions of the airway, due to the close anatomic association of the esophagus with the larynx, trachea, carina, and main bronchi. The esophagogram is invaluable in providing a clue to a malignant laryngeal or tracheal tumor and in identifying primary esophageal tumors that may secondarily invade the trachea. The presence, size, and course of a tracheoesophageal fistula can be established by an esophagogram.
Computed Tomography CT scanning is the preferred technique for evaluating the larynx, trachea, and main bronchi.15,17,18 Helical CT scanning has dramatically improved the quality of CT imaging of the airways by acquiring a volumetric data set in a single breath-hold while using a short scanning time. In comparison, conventional CT scanning uses a long scanning time and obtains individual axial scans during separate individual breath-holds. As a result, a major advantage of helical scanning is reduced cardiac and respiratory motion. The quality of two-dimensional (2-D) and three-dimensional (3-D) reformatted images are thus markedly improved. The
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4-6 Tracheomalacia. Lateral fluoroscopic images of the trachea during inspiration (A) and expiration (B), computed tomography (CT) of the midtrachea on inspiration (C) and expiration (D), and two-dimensional reformatted sagittal CT images of the trachea on inspiration (E) and expiration (F) demonstrate > 50% collapse in the anteroposterior diameter of the trachea, consistent with tracheomalacia. FIGURE
newest generation multidetector helical CT scanners employ multidetector arrays, which increase the speed of scanning by factors of 4, 8, or 16, thereby decreasing motion artifacts and improving the image quality of reformatted images. In addition, with multidetector helical CT imaging, high quality reconstructed
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images can be obtained routinely without prospective planning or rescanning the patient. The ability to create 2-D and 3-D reformatted images of the central airways overcomes the inherent limitations of axial images, including the limited ability of detecting subtle stenoses, evaluating the accurate craniocaudad extent of disease, visualizing obliquely oriented airways, and displaying the complex relationship of adjacent mediastinal structures (Figure 4-7A). Reformatted 2-D and 3-D images do not offer any new information; rather, they provide a complementary way to view the same data sets. Although the use of intravenous contrast is not necessary to assess the central airways, it is recommended to evaluate adjacent mediastinal extent of tumor and adenopathy or to assess adjacent mediastinal vascular structures or masses that may compress the airway. CT scanning of the airway is routinely obtained at end-inspiration during a breath-hold. When assessing tracheomalacia, it is helpful to obtain additional scan sequences at the same table levels during end-expiration. Reconstruction methods that are used for airway imaging include 2-D multiplanar and 3-D internal and external rendering techniques. 2-D images are the easiest to obtain because they can be generated at the CT console, and 2-D reformations can be displayed in the coronal or sagittal planes, orthogonal to a reference point or curved along the axis of the airway. 3-D reformations require the transfer of data to a separate workstation, but 3-D images of the airway can be visible on external 3-D rendered images or on internal renderings that create virtual bronchoscopic images of the central airways.
Magnetic Resonance Imaging Magnetic resonance imaging (MRI) is another modality being used for the radiologic evaluation of the larynx and trachea.19 A major advantage of MRI over CT is the acquisition of coronal, oblique, and sagittal sections that demonstrate long segments or the entire length of the trachea (Figure 4-7B). With MRI, it is possible to visualize the laryngeal and tracheal structures with great detail in transverse, sagittal, and coronal planes.3,20 Normal anatomic structures can be differentiated on the basis of different signal intensities on T1-weighted sequences. A bright signal is elicited by fat, hyaline cartilage, submucosal fascial planes
A FIGURE 4-7 Adenoid cystic carcinoma. A, Two-dimensional reformatted computed tomography image of the distal trachea and carina demonstrates a smooth intraluminal mass arising from the right lateral wall of the trachea. B, T1-weighted coronal magnetic resonance imaging through the trachea and carina in the same patient demonstrates a soft tissue mass in the distal trachea, consistent with adenoid cystic carcinoma.
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(paralaryngeal space), and false cords. Intermediate signal intensities are given off from the true cords, aryepiglottic folds, and the intrinsic laryngeal muscles. Calcified cartilage, the airway, and blood vessels produce low signals. MRI can be used in the assessment of tracheal stenosis and tumors and other paratracheal masses that compress the trachea (Figure 4-8). The examination is performed with a T1 sequence in the axial, coronal, and sagittal planes, supplemented by an axial T2 sequence. The T1-weighted sequence demonstrates the anatomy of the trachea and surrounding structures in great detail. The T2-weighted sequence adds to the characterization of the lesion, for example, differentiation of a cyst from a tumor. Carcinomas are characterized by intermediate signal intensities on the T1 sequences and high signal intensities on heavily T2-weighted images. On T1 sequences, the tumor may obliterate the high signal intensity areas (paralaryngeal and preepiglottic spaces), encroach on the signal void air spaces, and/or may cause erosion of the laryngeal or tracheal cartilaginous structures. MRI is the preferred method for evaluating paratracheal abnormalities in children because it does not involve ionizing radiation, and is particularly useful in studying vascular rings, slings, and dilated vessels that compress the trachea (Figure 4-9). In addition, MRI is the preferred modality in evaluating patients with paratracheal masses, who have contraindications to iodinated contrast material used for CT scans. Gadolinium, a MRI contrast agent, can be given safely to such patients. Dynamic MRI may also prove to be useful in evaluating tracheomalacia, although at the present time, inspiratory/expiratory CT scanning is still the examination of choice for studying this condition.
Congenital Abnormalities Tracheal Bronchus A tracheal bronchus is an anomalous bronchus that arises directly from the right side of the trachea.21,22 It is present in 0.25 to 1% of the human population. Most tracheal bronchi arise within 2 cm of the carina
4-8 Goiter. Coronal T1-weighted magnetic resonance image of the neck and chest reveals an enlarged thyroid gland causing marked tracheal narrowing at the thoracic inlet.
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FIGURE 4-9 Right-sided aortic arch. A, Anteroposterior view of the trachea demonstrates marked tracheal narrowing adjacent to the right aortic arch. B, Axial T1-weighted magnetic resonance imaging reveals compression of the midtrachea by a slightly dilated right-sided aortic arch.
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and are usually asymptomatic. Tracheal bronchi are classified into four different types: 1) rudimentary tracheal bronchus, typically arising as a blind outpouching of the right lower side of the trachea; 2) displaced bronchus, the most common anomaly in which one or two of the upper lobe segments are aerated by the tracheal bronchus; 3) supernumerary accessory bronchus, which arises from the trachea in addition to the normal right upper lobe (Figure 4-10); and 4) right upper lobe bronchus, with three normal segments arising above the tracheal bifurcation, sometimes a duplicate of a normal right upper lobe bronchus.
Congenital Tracheal Stenosis Congenital tracheal stenosis is a rare developmental abnormality of the trachea that may affect any or all parts of the trachea, in which the tracheal cartilages are hypoplastic, forming complete rings without a membranous posterior wall.23 As a result, the trachea is rigid and nondistensible. On CT scans, the trachea is narrowed, with identifiable calcified complete cartilaginous rings (Figure 4-11). Associated congenital anomalies are present in 80% of cases, including H-type tracheoesophageal fistula, laryngomalacia, subglottic stenosis, bronchial stenosis, hypoplasia or agenesis of the lungs, and other skeletal, cardiovascular, and intestinal anomalies.
Congenital Vascular Anomalies and Rings and Vascular Compression Vascular rings, slings, or mediastinal great vessels may compress the trachea and/or esophagus.24–27 In adults, most vascular compressions are related to acquired aneurysmal dilation of the great vessels. Contrast CT, magnetic resonance angiography, and/or angiography is indicated to establish a diagnosis. A barium swallow is necessary in the work-up of a child with respiratory symptoms, because the esophagus is frequently involved in compression syndromes.
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FIGURE 4-10 Tracheal bronchus. Computed tomography examination demonstrates a tracheal bronchus (A) arising from the distal trachea just above the carina (B). The normal right upper lobe bronchus is noted (C).
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Vascular rings are the most common symptomatic anomalies causing tracheal and esophageal compression and are classified as 1) double aortic arch or 2) right aortic arch with left ligamentum arteriosum, patent ductus, or aberrant left subclavian artery. The absence of an aortic arch on the left and the presence of a tracheal indentation by the right-sided aorta are critical findings. The distal trachea may be deviated to the left on the frontal view, and anteriorly on the lateral view. A double aortic arch causes both anterior and posterior compression of the trachea. A pulmonary artery sling occurs when an anomalous left pulmonary artery courses over the right main stem bronchus, near its origin from the trachea, and crosses posteriorly and to the left between the esophagus and the trachea, reaching the left hilum above the bronchus. As a result, it may compress the trachea and right main stem bronchus. A strong association exists with both tracheobronchial defects and cardiovascular anomalies. Diagnostic findings on barium swallow, CT, and MRI include indentation of the anterior aspect of the esophagus as well as tracheal deviation to the left just above the carina, by the aberrant pulmonary artery. The left hilum is situated more caudal than normal.27 Anterior compression of the trachea may occur by the innominate artery. The degree of compression is usually most severe in expiration. In children, the innominate artery often originates partially or totally to the left of the trachea and crosses in front of it, and clinical symptoms result because of crowding in the mediastinum. The lateral chest radiograph usually demonstrates a typical anterior compression of the trachea. The esophagus is usually not affected on an esophagogram.26
Postpneumonectomy Syndrome Postpneumonectomy syndrome is a rare condition that occurs in children or adults following a pneumonectomy.28,29 For reasons that are not well understood, in certain patients, the heart and mediastinum shift excessively toward the side of the pneumonectomy and the great vessels rotate significantly. In the case
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4-11 Congenital tracheal stenosis. A, Coronal tomogram demonstrates diffuse stenosis of the intrathoracic trachea. B, Computed tomography image through the midtrachea reveals a complete cartilaginous tracheal ring and significant stenosis of the trachea (C). D,E, Following a slide tracheoplasty procedure, the tracheal lumen is significantly greater. FIGURE
of a right pneumonectomy with a left-sided aortic arch, the remaining distal trachea and/or left main bronchus becomes interposed between the pulmonary artery anteriorly and the aorta and thoracic spine posteriorly, resulting in a stenosis of the distal trachea or main bronchus (Figure 4-12). A similar compli-
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4-12 Postpneumonectomy syndrome. Posteroanterior (A) and lateral (B) radiographs of the chest following a right pneumonectomy reveal marked shift of the mediastinum and heart to the right and posteriorly. A contrast-enhanced chest computed tomography scan reveals counterclockwise rotation of the pulmonary artery (P) that is horizontal in orientation (C, D). The left main bronchus (arrow) is narrowed and interposed between the left main pulmonary artery (PA) anteriorly and the descending aorta (A) posteriorly (D, E). FIGURE
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cation may be seen following a left pneumonectomy with a right-sided aortic arch. Cross-sectional imaging with CT and/or MRI is essential to establish the vascular and bronchial relationships and to define the position and extent of the tracheobronchial obstruction. Inspiratory/expiratory CT imaging is useful to identify a malacic segment that may rarely occur at the level of the stenosis. Postoperative imaging following repositioning of the mediastinum helps to redefine the vascular and tracheobronchial positions and confirm establishment of a patent airway.
Trauma Larynx Laryngeal trauma can be caused by external or internal injuries. External injuries are the result of blunt or penetrating trauma. Internal injuries are usually caused by prolonged intubation or chemical or thermal burns.30 Trauma is characterized by mucosal disruption of soft tissues, swelling, and collection of air in the soft tissue structures of the larynx including neck and mediastinum. These soft tissue injuries may be associated with cartilaginous fractures, and dislocations of the arytenoids and epiglottis.31 The blood and edema fluid insinuate along the deep spaces of the larynx, predominantly in the paralaryngeal and epiglottic spaces, followed by a variable degree of airway narrowing. Fractures of the thyroid cartilage may be transverse or vertical, resulting in hemorrhage into the preepiglottic space, with consequent posterior displacement of the epiglottis. Fractures in the cricoid cartilage are often vertical and lead to a variable degree of disruption of the signet ring. Fractures of the cartilaginous structures are associated with a variable degree of displacement and hematoma formation. The arytenoids may be displaced as an isolated incidence or in conjunction with fractures of the cartilaginous structures. They are most often displaced anteriorly and superiorly. Vocal cord motion is usually impaired as a result of hematoma formation, fractures, arytenoid dislocation, fibrosis, or recurrent laryngeal nerve paresis. Disruption of the cricothyroid joint may occur and also lead to dysfunction of the vocal cords. The sequelae of severe laryngeal injuries are variable degrees of stenosis, which may involve the entire larynx or may be localized to the supraglottic, glottic, or subglottic larynx. CT is the modality of choice for demonstrating the various described findings in laryngeal trauma.32 CT will also demonstrate the extent of soft tissue edema, hematoma formation, the location and extension of fractures, and the deformity of cartilaginous structures after healing (Figure 4-13).
Trachea and Main Bronchi Tracheobronchial rupture is a rare injury that results from a decelerating injury. The possible mechanisms of injury include compression of the airway between the sternum and the vertebral column, a sudden deceleration of pendulous lung with a fixed trachea creating shearing forces, and forced expiration against a closed glottis raising intrabronchial pressure. The site of airway rupture and its extent determine the radiographic findings. The tracheal or bronchial tear is usually complete and will lead to subcutaneous emphysema, pneumomediastinum, or pneumothorax.33 Tears of the trachea and proximal left main bronchus generally leave the parietal pleura intact and will result in pneumomediastinum (Figure 4-14). Distal left main bronchial tears and right main bronchial tears will generally communicate with the pleural cavity and result in pneumothorax (Figure 4-15). Tears generally occur within 2.5 cm of the carina, and are more commonly seen on the right side. If the central anchoring components of the lung are completely ruptured and the main bronchus disrupted, the lung may collapse peripherally from the hilum, in what has been described as the “fallen lung” sign. In some cases, there may be an incomplete tear and an absence of an air leak when the integrity of the peribronchial or peritracheal connective tissue is maintained, when a cuff occludes the tear, or when fibrin seals the tear. Tears that present in this fashion are often missed initially and have a delayed presentation (Figure 4-16). If healing develops, an
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FIGURE 4-13
Laryngeal trauma. Axial computed tomography section through the subglottic space of the larynx defines a fracture in the anterior thyroid cartilage (arrow).
B FIGURE 4-14 Acute tracheal tear. A, Posteroanterior chest radiograph demonstrates marked diffuse pneumomediastinum extending into the neck. B, Computed tomography scan through the upper chest reveals extensive pneumomediastinum and subcutaneous emphysema. There is a tear of the membranous wall of the trachea.
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FIGURE 4-15 Acute left main bronchial rupture. Computed tomography scans through the carina (A) and left main bronchus (B) reveal extensive pneumomediastinum, subcutaneous emphysema, and a leftsided pneumothorax. There is marked narrowing of the left main bronchus at the site of laceration (B).
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untreated partial laceration will develop a stenosis with a typical hourglass configuration and potentially cause distal collapse of a lung or lobe (Figures 4-17, 4-18). Tracheobronchial trauma is often associated with aortic injury and fractures of the first three ribs or sternum.34–36
Traumatic and Postintubation Stenosis One of the complications to the larynx and trachea, most commonly seen following intubation, is granuloma formation. Granulomas occur primarily in the posterior supraglottic, glottic, and subglottic larynx, and cervical trachea (Figures 4-19, 4-20). They can attain a large size with subsequent airway obstruction. Late changes secondary to trauma represent laryngeal stenoses causing airway obstruction. These stenoses may occur in the supraglottic, glottic, and subglottic larynx,37 or as a combination of all the sites with extension into the cervical trachea (Figures 4-21, 4-22, 4-23).38 In severe cases, the entire larynx and/or trachea may become obliterated. Tracheal stenosis is also encountered post-tracheostomy and postintubation.39 They occur primarily at two sites: 1) the tracheostomy opening or stoma, and 2) in the area of the balloon cuff. In a small percentage of cases, both lesions will occur simultaneously.40 With the introduction of compliant, extensible, large-volume latex cuffs, this complication has been prevented to a great degree, and, consequently, the incidence of postcuff stenosis has markedly decreased.
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FIGURE 4-16 Subacute right main bronchial tear. A, Anteroposterior chest radiograph reveals complete opacification of the right hemithorax and shift of the trachea and mediastinum to the right, consistent with right lung collapse following contained laceration of the right main bronchus. Computed tomography scans with lung windows at the carina (B) and slightly below the carina (C) demonstrate narrowing of the lacerated right main bronchus. D,E, Soft tissue windows at the same levels reveal a fluid-filled, displaced distal right main bronchus (arrow).
At the stomal site, a large stoma, superimposed infection, or the use of rigid connecting systems increases the incidence of stomal strictures due to pressure erosion.41 The stenosis at the tracheostomy stoma frequently involves the anterior and lateral tracheal wall, and forms a triangularly-shaped area of narrowing (Figure 4-24). Changes in the tracheal wall consist of fibrosis, often associated with granulation tissue. In many instances, a variable amount of calcium and bone that are deposited in the tracheal wall are readily demonstrated on the CT scan. Another complication seen at the tracheostomy site is a formation of an anterior tracheal wall flap from above the stoma, caused by inversion of the anterior tracheal wall into the adjacent lumen. Granulation tissue may form on the flap and increase the severity of the obstruction.
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FIGURE 4-17 Post-traumatic tracheal stenosis. A, Computed tomography scan with intravenous contrast material demonstrates a narrowed, misshapen trachea surrounded by soft tissue density, consistent with fibrosis at the site of healed tracheal rupture. B, A lung window through the superior mediastinum reveals marked tracheal stenosis.
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4-18 Missed left main bronchial stenosis due to missed bronchial rupture. Posteroanterior chest radiograph (A) and anteroposterior tomogram (B) reveal collapse of the left lung distal to a left main bronchial stenosis.
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FIGURE 4-19 Subglottic granuloma. Lateral neck view illustrates a sharply delimited polypoid mass in the anterior subglottic space, consistent with a granuloma (asterisk).
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FIGURE 4-20 Tracheal granuloma adjacent to a tracheostomy canula. Oblique spot film demonstrates a granuloma at the tracheostomy tube (arrows).
FIGURE 4-21 Subglottic stenosis. Axial computed tomography scan demonstrates diffuse circumferential thickening of the submucosa in the subglottic space (asterisk). Note the normal cricoid cartilage (arrow).
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FIGURE 4-22 Infraglottic and adjacent cervical tracheal stenosis. Lateral view of the neck illustrates slight narrowing of the inferior portion of the subglottic space (asterisk) and adjacent trachea posteriorly. There is thickening of the tracheal wall with some calcification (arrows).
4-23 Subglottic and cervical tracheal stenosis. Lateral neck view shows a long severe subglottic and cervical tracheal stenosis (arrow). Note the tracheostomy tract (asterisk). The inferior margin of the larynx is indicated by a dot. FIGURE
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FIGURE 4-24 Stomal stenosis of the cervical trachea. A, Anteroposterior high-kilovoltage view outlines a localized stenosis in the cervical trachea (arrows). B, Coronal tomogram shows the polypoid configuration of the stenosis. C, Lateral tomogram shows the stenosis chiefly anteriorly and laterally.
When the tracheostomy tube is removed, it may act as a ball-valve mechanism and obstruct the tracheal lumen. These flaps are optimally demonstrated on lateral neck films as a variable and sometimes mass-like soft tissue density above the stomal opening. A cuff stenosis occurs 1 to 2 cm below the tracheal stoma and is circumferential in configuration. Inflammatory histologic changes are noted within 24 to 48 hours following intubation. The inflammation leads to superficial tracheitis and mucosal ulceration within 1 week. Deeper mucosal ulceration may develop, along with exposure of the underlying cartilage, in 1 to 3 weeks. If the inflammatory process is not halted, cartilage will be exposed and chondritis will ensue, with fragmentation and eventual total destruction of the cartilaginous supporting structures in a period of 2 to 3 weeks following the intubation. Reparative healing will supersede the inflammatory process and lead to fibrotic change and formation of granulation tissue with tracheal narrowing.40 The length and severity of the stenosis are related to the pressure within the cuff, size and shape of the cuff, the number of days of intubation, and the peak inspiratory pressure. The stenosis is usually circumferential and from 1 to 4 cm in length. In its fully developed stage, the lesion at the cuff site may vary in severity, from a circumferential diaphragm of fresh granulation tissue to dense rings of mature fibrous tissue partially covered by metaplastic squamous epithelium, extending over a variable length. A preliminary high-kilovoltage oblique film of the trachea defines the level and length of the stenosis (Figure 4-25). For detailed assessment of the lumen, thickness of the tracheal wall, and presence of mural calcification, CT is currently indicated, whereas in previous years, tomography was performed (Figures 4-26, 4-27). Tracheomalacia develops, in a small percentage of cases, above the cuff stenosis site or at the stomal site. When the full thickness of the tracheal wall has been damaged, so that cartilages are no longer present in the area of injury, malacia may ensue. In a small number of cases, a malacic segment alone may be found at the cuff site. The malacia can be demonstrated by fluoroscopic examination with observation of the trachea during coughing and maximal inspiration and expiration. There is collapse of the entire tracheal wall, especially the anterior wall. Tracheal stenosis may be associated with other findings and complications, such as vocal cord paralysis, and infraglottic stenosis. Subglottic stenosis is usually secondary to damage of the cricoid cartilage from the cuff of an endotracheal tube, or a high tracheostomy tube with erosion of the
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FIGURE 4-25 Cuff stenosis of the cervical trachea. A, Anteroposterior high-kilovoltage view demonstrates narrowing of the trachea (arrow). B, Tomographic image reveals circumferential narrowing of the lumen. Note the asterisk in the subglottic space of the larynx.
cricoid, or from a cricothyroidotomy. In a small number of cases, tracheoesophageal fistula may develop, followed by a sudden increase in profuse secretions plus food aspiration.42 These fistulas are best illustrated with a barium swallow. Different types of foreign bodies may lodge in the trachea, especially in young children, followed by recurrent obstructive pneumonitis, if undetected by either radiographic means or bronchoscopy. Foreign body location in the larynx is uncommon but has occasionally been encountered in adults (Figure 4-28).
Saber-Sheath Trachea Saber-sheath trachea is a condition closely related to chronic obstructive lung disease, especially chronic bronchitis.43,44 In this condition, there is coronal narrowing and sagittal widening of the intrathoracic trachea, with a tracheal index (coronal/sagittal diameter) < 0.5 at the level of the aortic arch. It has been theorized that repeated excessive tracheal collapse during chronic coughing leads to degenerative softening, revascularization, and ossification of the tracheal cartilages, resulting in a fixed coronal narrowing. Radiologically, there is a normal circular cervical tracheal configuration with an abrupt transition to a saber-sheath configuration, beginning at the thoracic inlet and extending to the carina (Figure 4-29). The tracheal cartilages may ossify in this condition, but the trachea otherwise maintains a smooth outline. Tracheomalacia is not a recognized feature.
Granulomatous Lesions The surface contour of the endolarynx is smooth and symmetrical in inflammatory conditions, with the exception of granulomatous diseases. There is usually preservation of mobility of intralaryngeal structures,
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FIGURE 4-26 Cuff stenosis of the cervical trachea. A, Coronal tomogram delineates circumferential narrowing of the trachea. B, Anteroposterior high-kilovoltage view illustrates narrowing of the lumen (arrows).
although slight limitation may occur. Chronic granulomatous lesions of the larynx display a diffuse or localized nodular soft-tissue thickening. Frequently, a malignant tumor cannot be differentiated from the granulomatous process, and a biopsy is often mandatory for a definitive diagnosis. Granulomatous processes may extend from the subglottic part of the larynx into the cervical trachea.
Tuberculosis Larynx. Tuberculosis of the larynx is usually secondary to pulmonary tuberculosis and commonly affects the posterior structures of the larynx. Diffuse swelling or a localized irregular mass may be found, depending on whether the pathologic process is acute, exudative, or chronic productive.45 Tracheobronchial. Tuberculous tracheobronchial stenosis may be caused by extrinsic compression or by adjacent lymphadenopathy or by granulomatous changes within the airway (Figure 4-30). In the hyperplastic stage, tubercles form in the submucosal layer, and ulceration and necrosis of the wall ensues. In the fibrostenotic stage, a smooth stenosis is formed.46 Radiographically, in the hyperplastic stage, the tracheobronchial walls will be nodular and thickened with variable degrees of stenosis (Figure 4-31). Associated lymphadenopathy may demonstrate rim enhancement with intravenous contrast. There may be parenchymal
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4-27 Cuff stenosis of the cervical trachea. A, Coronal tomographic section reveals a localized well-defined cuff stenosis (arrow). B, Lateral view of the cervical trachea demonstrates the stenotic area, chiefly posteriorly (arrow). FIGURE
4-28 Chicken bone in the larynx. Lateral neck view demonstrates a chicken bone in the larynx (arrows).
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FIGURE 4-29 Saber-sheath trachea. Posteroanterior (A) and lateral (B) chest radiographs demonstrate narrowing of the trachea in the coronal plane and widening in the sagittal plane.
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FIGURE 4-30 Tracheobronchial tuberculosis with lymphadenitis. Computed tomography scan at the level of the carina with soft tissue windows (A) and lung windows (B) reveals an enlarged necrotic low attenuation lower paratracheal lymph node that contains calcification (A). There is extension of the granulomatous process narrowing the right main bronchus (B).
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4-31 Tuberculous bronchial stenosis, hyperplastic stage. Computed tomography scan demonstrates thickening and nodularity of the right main and right upper lobe bronchi associated with right hilar adenopathy (A) and cavitation of the right lower lobe (B).
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cavitation within the lobes drained by the affected bronchi. In the fibrostenotic stage, the bronchi remain thickened, but have a smooth luminal stenosis (Figure 4-32). If the stenosis is complete, total collapse of a lobe or entire lung will be present. Broncholithiasis is a late sequela of tuberculosis or histoplasmosis. Rarely, calcified mediastinal nodes will erode into a bronchus, causing obstructive complications such as atelectasis, repeated pneumonia, or bronchiectasis. CT is useful to identify the presence and extent of the obstructing bronchial lesion (Figure 4-33). Calcification within the broncholith and associated mediastinal lymph nodes helps to establish the diagnosis.
Fungal Disease Fungal diseases, such as blastomycosis, candidiasis, histoplasmosis, mucormycosis, rhinosporidiosis, and coccidioidomycosis, produce radiographic intrinsic stenosis of the central airways similar to those described with tuberculosis (Figure 4-34).
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4-32 Tuberculous stenosis of the trachea and main bronchi, fibrotic stage. A, Chest radiograph reveals collapse of the left lung. Computed tomography scans reveal diffuse smooth stenosis of the trachea (B) and main bronchi (C). There is collapse of the left lung and a calcific left fibrothorax. FIGURE
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Fibrosing mediastinitis is a complication of granulomatous mediastinitis, resulting from infection from Histoplasma capsulatum and, less commonly, Mycobacterium tuberculosis. It has also been associated with the use of methysergide. Fibrosing mediastinitis is characterized by a proliferation of fibrous tissue in the mediastinum, which surrounds, invades, and sometimes obliterates normal structures, including the trachea and bronchi, esophagus, vena cava, pulmonary veins and arteries, and thoracic duct. Calcification is often present within the mediastinal fibrosis, identifiable on chest radiographs and CT (Figure 4-35). Contrast enhanced CT or MRI is useful to evaluate vascular invasion or occlusion by the fibrosis.47,48
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4-33 Tuberculous broncholithiasis. The posteroanterior chest radiograph (A) and anteroposterior tomogram (B) reveal collapse of the right upper lobe distal to a calcified right upper lobe-filling defect, consistent with a broncholith. C, A computed tomography scan reveals complete obstruction of the right upper lobe bronchus by a calcified endobronchialfilling defect associated with a densely calcified right paratracheal lymph node in a patient with healed tuberculosis. FIGURE
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Sarcoidosis Sarcoidosis of the larynx and trachea is characterized by diffuse or nodular thickening, and in some cases, a tumor-like infiltration. The epiglottis is most frequently involved. Other airway involvement includes tracheobronchial mural thickening, which may be smooth, irregular, or nodular luminal narrowing (Figure 4-36). Airway compression by lymphadenopathy may occur. Thickening of the tracheobronchial wall represents the presence of granulomas in the bronchial mucosa and along the bronchovascular interstitium, accounting for the high diagnostic success of transbronchial biopsy (Figure 4-37).49
Scleroma Scleroma is caused by Klebsiella rhinoscleromatis, a gram-negative bacterium. It is a chronic granulomatous disorder that commonly involves the subglottic larynx and cervical trachea and is characterized by granu-
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4-34 Bronchial stenosis due to mucormycosis. A computed tomography scan reveals nodular stenosis of the right upper lobe and main bronchi. There is marked cavitation of the right upper lobe by the infection.
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FIGURE 4-35 Fibrosing mediastinitis. Computed tomography scans through the distal trachea (A) and main bronchi (B) with intravenous contrast reveal narrowing and distortion of the trachea and bronchi. There is surrounding fibrosis that contains calcification.
lomatous masses or nodular and diffuse infiltrations. Although the proximal trachea is most commonly involved, the entire trachea and even the main bronchi may be affected by obstruction. The radiologic findings correlate with the three clinical stages of inflammation: 1) the catarrhal stage, 2) the granulomatous proliferative stage, and 3) the sclerotic cicatricial stage.50
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4-36 Tracheal stenosis in end-stage sarcoidosis. Posteroanterior (A) and lateral (B) chest radiographs reveal marked tracheal and bronchial stenosis and distortion. There is marked pulmonary fibrosis and traction bronchiectasis related to pulmonary sarcoidosis.
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FIGURE 4-37 Diffuse tracheobronchial stenosis in sarcoidosis. Computed tomography scans through the main bronchi (A) and left upper lobe bronchus (B) reveal diffuse bronchial wall thickening and calcification. Calcified subcarinal lymph nodes are noted.
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Wegener’s Granulomatosis Wegener’s granulomatosis may involve the upper and lower respiratory tract, usually in conjunction with renal and other organ involvement.51 Wegener’s granulomatosis may involve the subglottic larynx and cause diffuse narrowing of the subglottic airway (Figure 4-38). Diffuse tracheobronchial involvement is rare and usually presents late in the disease process. Radiographic findings include tracheobronchial narrowing, thickening, and irregularity that may be focal or diffuse (Figures 4-39, 4-40). Occasionally, granulomatous tissue can obstruct a bronchus, causing atelectasis. Mediastinal and/or hilar adenopathy may be present on CT.
Other Benign Infiltrative Lesions Idiopathic Laryngotracheal Stenosis Idiopathic laryngotracheal stenosis is a rare cause of narrowing of the larynx and cervical trachea that typically affects middle-aged women who have no history of trauma, infection, or systemic disorder. The stenotic areas show dense keloid fibrosis involving the adventitia and the lamina propria, sparing the mucosa, muscularis propria, and the cartilages. The radiographic appearance is variable, including smooth and tapered, or irregular, lobulated, and eccentric lesions that are 2 to 4 cm in length (Figure 4-41).52
Relapsing Polychondritis Relapsing polychondritis is an uncommon multisystem disease causing progressive, episodic inflammation and destruction of hyaline cartilage and other tissues. Clinical manifestations include auricular chondritis, arthritis, nasal chrondritis, ocular inflammation, respiratory tract involvement, audiovestibular damage, cardiovascular involvement, and skin disease. Respiratory tract involvement is the major cause of death. Relapsing polychondritis thickens the laryngotracheal cartilages, causing airway narrowing adjacent to the epiglottis, aryepiglottic folds, glottis, subglottis, and upper trachea. Progressive thickening of the distal trachea and bronchi may develop, but this is less common. There is a spectrum of findings in the airways.53,54 In the early stages of the disease, mucosal inflammation is present, causing thickening and narrowing of the central airways. As the disease progresses and
4-38 Wegener’s granulomatosis subglottic larynx. Axial computed tomography section shows diffuse soft tissue thickening of the subglottic larynx (asterisk) adjacent to the cricoid cartilage (arrow). FIGURE
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FIGURE 4-39 Tracheal stenosis due to Wegener’s granulomatosis. An oblique tomogram of the trachea reveals a diffuse tracheal stenosis with an hourglass configuration.
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4-40 Wegener’s granulomatosis of the main bronchi. Computed tomography scans through the main bronchi reveal diffuse nodularity of the bronchial walls.
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FIGURE 4-41 Idiopathic tracheal stenosis. Anteroposterior tracheal tomograms in two separate patients reveal subglottic stenosis with a diffuse smooth stenosis (A) and a mass-like obstructing lesion of the proximal trachea (B).
cartilage is destroyed, the airways become collapsible and tracheobronchomalacia develops. Once the destroyed cartilages are replaced by fibrous tissue, a diffuse fixed stenosis develops (Figure 4-42). In such cases, CT demonstrates diffuse smooth thickening of the involved trachea and bronchi (Figure 4-43). Occasionally, calcification is seen within the thickened walls.
Tracheopathia Osteochondroplastica Tracheopathia osteochondroplastica is a rare benign condition manifested by multiple hard osteocartilaginous masses in the submucosa of the anterior and lateral walls of the cervical and intrathoracic trachea and main bronchi (Figure 4-44). The posterior membranous wall is typically uninvolved, a finding that distinguishes this disease entity from other diffuse infiltrating diseases. The size and distribution of the nodules are best depicted by CT. Submucosal nodules may range in size from a few millimeters to larger obstructing nodules. Ossification within the nodules can be identified on CT scans. The tracheobronchial wall is characteristically rigid on inspiratory/expiratory CT scans and fluoroscopy.55
Amyloidosis Tracheobronchial amyloidosis results from the extracellular deposition of an insoluble protein that stains with Congo red. Airway involvement is most commonly seen in the primary form of the disease. Although any portion of the airway can be involved, distal tracheal and bronchial involvement is more common. Amyloidosis of the airway may be focal or mass-like, mimicking a tumor, or may cause diffuse thickening of the tracheobronchial wall.56
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4-42 Relapsing polychondritis of the glottis and proximal trachea. An anteroposterior tomogram of the proximal trachea and larynx reveals a smooth subglottic stenosis.
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A 4-43 Relapsing polychondritis. Computed tomography scans demonstrate diffuse thickening of the trachea (A) and bronchial (B) cartilages. FIGURE
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FIGURE 4-44 Tracheopathia osteochondroplastica. A, Anteroposterior tomogram reveals diffuse nodularity of the trachea and bronchi. B, Computed tomography scan at the level of the midtrachea demonstrates thickening and nodularity of the tracheal cartilage, sparing the membranous wall.
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Radiographs and CT scans demonstrate tracheobronchial wall thickening and nodularity or, less commonly, a focal endoluminal lesion (Figures 4-45, 4-46). The airway abnormality may calcify and focal lesions may demonstrate intense vascular enhancement with intravenous contrast material on CT, or gadolinium on MRI (Figure 4-47). Associated mediastinal or hilar adenopathy is sometimes present and may contain calcification.
Benign Tumors and Cysts Benign tumors of the trachea and larynx are very rare and account for 10% of tracheal tumors.57,58 Ninety percent of tumors which are encountered in the pediatric age group are benign. Benign tumors represent a large group of diverse lesions that manifest as sharply-defined masses with no invasive characteristics.20,59 They are usually hom*ogeneous, and the majority of lesions have no characteristic features to differentiate them by radiologic means.60
Papillomas Papillomatosis is the result of a multicentric viral infection with the human papilloma virus. Papillomas occur either singly or as multiple, irregular tumor excrescences that generally arise from the true vocal cords.61 This tumor most often involves the superior surfaces or free margins of the vocal cords and, less commonly, occurs in the supraglottis and subglottis (Figure 4-48). Papillomas are divided into juvenile and adult groups, where those of the juvenile group often manifest as multiple lesions, most commonly found in the larynx. The papillomas may recur or may spread diffusely through the trachea, bronchi, and lungs following excision. In the lungs, sheets of squamous cells proliferate within alveoli, forming nodules that characteristically cavitate. The adult type of lesion often presents as a solitary mass, with a lesser propensi-
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4-45 Diffuse tracheobronchial amyloidosis. Computed tomography scans at the level of the cricoid cartilage (A), midtrachea (B), and main bronchi (C) reveal diffuse tracheobronchial thickening and narrowing, with calcification of the airway walls. FIGURE
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ty to recur after removal. Transformation of the lesions into invasive squamous cell carcinoma is well known. Radiographically, the tracheal walls may appear thickened and nodular in appearance, either in a focal or diffuse fashion. These are sometimes visible on chest radiographs, but are best demonstrated by CT (Figure 4-49). Multiple pulmonary nodules with and without cavitation can be identified on chest radiographs and CT (Figure 4-50).
Chondromas Chondromas of the larynx are uncommon lesions that occur most frequently in middle-aged men. The tumor is smooth, submucosal, and firm in consistency.62 On radiographic and CT examination, the tumor presents as a sharply-defined mass and frequently contains mottled calcifications (Figure 4-51). Radiologically, a chondroma cannot be distinguished from a chondrosarcoma; this distinction may also be difficult on histopathologic examination. The most common location is the inner surface of the cricoid lamina (70%) (Figure 4-52). Less often, chondromas arise from the thyroid, arytenoid, or epiglottic cartilages. On rare occasions, chondromas may be situated on the upper surfaces, free margins, or
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undersurfaces of the vocal cords. Chondromas may occur in the trachea and bronchi but do so less commonly than laryngeal chondroma.
Hemangiomas Hemangiomas are uncommon lesions in the larynx and occur even less commonly in the trachea. The pediatric type of hemangioma usually manifests by 6 months of age, is subglottic in location, and causes signs of airway obstruction.63 Typically, these children exhibit hoarseness, stridor, and dysphagia with poor feed-
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B 4-46 Focal amyloidosis of the left main bronchus. A, The posteroanterior chest radiograph demonstrates a nodularfilling defect in the distal trachea obstructing the left main bronchus with distal left lung atelectasis. B, Computed tomography (CT) scan without intravenous contrast reveals a calcified soft tissue mass obstructing the left main bronchus. C, A CT scan with intravenous contrast demonstrates enhancement within the obstructing left main bronchial lesion. FIGURE
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FIGURE 4-47 Focal tracheal amyloidosis. A, Anteroposterior tracheal tomogram reveals a large calcified soft tissue mass nearly obstructing the proximal trachea. B, Computed tomography scan without contrast reveals marked calcification within the mass and extension of the mass into the right paratracheal soft tissues. C, T1-weighted magnetic resonance image of the proximal trachea with gadolinium reveals marked enhancement of the mass.
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ing. In the anteroposterior radiographic film, there is subglottic and often asymmetrical narrowing, and evidence of a distinctive, hom*ogeneous, sharply-defined soft tissue mass. In adults, laryngeal hemangiomas usually arise in the supraglottic larynx as a sharply-defined, hom*ogeneous mass that may contain phleboliths. Most of the reported cases are of the cavernous variety, as opposed to capillary hemangiomas, which are seen in infancy.
Other Benign Tumors Miscellaneous benign tumors are encountered in the larynx and trachea. These include neurogenic tumors, pleomorphic adenoma, oncolytic tumor, granular cell tumor, paraganglioma, lipoma, fibrous histiocytoma, rhabdomyoma, and hamartomas (Figure 4-53).64–72 Conventional radiographs do not usually reveal any characteristic features that allow a specific histopathologic diagnosis. CT can identify fatty attenuation
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within hamartomas and lipomas and increased contrast enhancement within paragangliomas. A chondroid matrix can be identified within chondromas and chondrosarcomas.
Laryngoceles Various cystic lesions are encountered in the larynx that are either retention cysts or laryngoceles. Laryngoceles are air-filled or fluid-filled outpouchings of the mucosa of the laryngeal ventricles. They extend from the ventricle into the adjacent aryepiglottic fold and are then referred to as internal laryngoceles.73,74 If the air-filled structure herniates through the thyrohyoid membrane, external laryngoceles result, which are usually well defined. Intralaryngeal expansion leads to a variable degree of airway obstruction, depending on the size of the lesion. If these laryngoceles are filled with fluid, they manifest as hom*ogeneous, dense masses that cannot be differentiated from a benign tumor.
Tracheal Cyst or Tracheocele A tracheal cyst is a thin-walled air-containing paratracheal cavity that is visible on chest radiographs and chest CT scans (Figure 4-54). The tracheal cyst is a circ*mscribed saccular tracheal outpouching of the posterior wall of the trachea. The cyst may rarely contain an air–fluid level. Dynamic bulging can be observed during a Valsalva maneuver at fluoroscopy or on CT. This condition occurs through a localized weakness of the membranous part of the trachea and is theorized to be associated with obstructive lung disease.75
B FIGURE 4-48 Squamous papilloma of the larynx. A, Lateral view of the neck illustrates a polypoid irregular mass in the supraglottic, glottic, and subglottic portions of the larynx (arrows). B, Axial computed tomography scan outlines the lesion anterolaterally on the right, with erosion of the adjacent cartilage.
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FIGURE 4-49 Tracheal papillomatosis. Posteroanterior chest radiograph (A) and anteroposterior tomogram (B) demonstrate diffuse nodularity of the trachea. Computed tomography scans with soft tissue windows (C) and lung windows (D) demonstrate multiple tracheal wall papillomas that partially obstruct the lumen.
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Diffuse papillomatosis. A, Lateral tomogram of the larynx and cervical region reveals diffuse nodularity throughout the larynx and proximal trachea, with partial obstruction of the lumen. B, A chest computed tomography scan reveals small pulmonary nodules and a cavitary nodule in the right lower lobe, consistent with pulmonary papillomatosis. Posteroanterior (C) and lateral (D) chest radiographs demonstrate bilateral pulmonary nodules, some of which are cavitary.
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FIGURE 4-51 Chondroma of the larynx. Axial computed tomography scan shows a calcified mass arising from the right thyroid cartilage and adjacent cricoid cartilage. The tumor bulges into the airspace and displaces the thyroid cartilage laterally.
4-52 Chondroma. Lateral tomogram of the larynx (A) and computed tomography scans (B, C) through the cricoid cartilage reveal a calcified mass arising from the cricoid cartilage, consistent with a chondroma.
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FIGURE 4-53 Granular cell myoblastoma of the cervical trachea. Lateral view of the neck outlines an oval-shaped, sharply-defined soft tissue mass arising from the posterior cervical tracheal wall.
B 4-54 Tracheal cyst. Anteroposterior tomogram (A) and computed tomography scan of the upper trachea (B) demonstrate a thin-walled air-filled right paratracheal cyst. FIGURE
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Malignant Lesions Epithelial Tumors Larynx. The majority of laryngeal malignancies (90%) represent epithelial neoplasms. Among these, 50 to 70% of laryngeal cancers are glottic in nature, 30 to 35% represent supraglottic carcinomas, and 4 to 6% represent subglottic carcinomas.76,77 Tumor size, location, and histologic grading are important parameters that determine the occurrence of lymph node metastases. Metastatic lymph nodes are more common when the primary tumor in the supraglottic larynx is greater than 2.0 cm in diameter and is poorly differentiated. Supraglottic carcinomas arise from the laryngeal surface and rim of the epiglottis, aryepiglottic folds, arytenoids, false cords, and laryngeal ventricles.78 They commonly extend across the midline, invade the extralaryngeal structures by direct extension to the pyriform sinuses, extend to the postcricoid region, and potentially extend to the cervical esophagus, valleculae, and base of tongue. Vocal cord cancers arise commonly from the anterior two-thirds of the vocal cords and may spread via the anterior commissure to the subglottic space (20%) and infrequently into the cervical trachea (Figure 4-55).77 Deep penetration of the cord by tumor into the vocalis muscle causes fixation of the cord. Carcinomas arising in the cervical trachea may involve by superior extension the subglottic larynx. In larger carcinomas, it is often difficult to determine whether the origin is from the cervical esophagus, the cervical trachea, or an extension of a subglottic carcinoma into the upper trachea. Stomal recurrence postlaryngectomy is encountered in 5 to 15% of cases. The tumor manifests as single or multiple nodules, at or near the stomal margin, involving the skin or tracheal mucosa. Deep invasion is commonly present, associated with ulceration at the skin margin. CT evaluation provides valuable information concerning extension of malignant tumor to the following areas: 1) the anterior commissure, 2) the paracordal and para-arytenoidal areas, 3) the preepiglottic and subglottic spaces, 4) cartilage invasion (see Figure 4-55), extralaryngeal extension of an endolaryngeal tumor, and 6) extension of pyriform sinus carcinomas through the cricothyroid space to involve the postcricoid region and cervical esophagus.78 Contrast CT or MRI is used to evaluate the tracheal extension and tumor invasion of the upper mediastinum prior to surgery and/or radiation therapy.79–81
4-55 Vocal cord carcinoma (left). Axial computed tomography section shows a left vocal cord carcinoma causing cartilaginous erosion and extension into adjacent soft tissue structures.
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Trachea. Although very rare, primary malignancies of the trachea are much more common than benign tumors.82 Squamous cell carcinoma is the most common tracheal malignancy (50%), followed by adenoid cystic carcinoma (30%) and adenocarcinoma (10%). Other less frequently encountered malignancies include mucoepidermoid carcinoma, undifferentiated carcinoma, small cell carcinoma, and carcinosarcoma.1,83 SQUAMOUS CELL CARCINOMA. Squamous cell carcinoma may present as a focal mass with a tendency for exophytic growth and a propensity to invade the mediastinum (Figure 4-56).84 Synchronous and metachronous squamous cell carcinomas of the larynx, lungs, and esophagus are found in many patients. CT is useful in demonstrating the primary tumor and its extent in the trachea and adjacent mediastinum, as well as associated adenopathy within the mediastinum and hilum. ADENOID CYSTIC CARCINOMA. Adenoid cystic carcinoma tends to grow with endophytic spread in the submucosal plane of the trachea and bronchi (Figure 4-57).85,86 On radiographs and CT and MRI scans, the trachea appears thickened with a smooth nodular appearance, associated with luminal narrowing (Figure 4-58). The tumor may extend into the adjacent soft tissues of the neck and mediastinum, depicted on CT or MRI as extension into the adjacent mediastinal fat. Regional lymph nodes in the neck and mediastinum are the first to be involved by metastases. Hematogenous metastases to lung, bones, and liver occur later in the disease progression. MUCOEPIDERMOID TUMORS. Mucoepidermoid tumors are very uncommon tumors of the trachea, central bronchi, and rarely of the lung.87 These tumors may be of either high-grade or low-grade malignancy. The radiographic findings are of a focal endoluminal soft tissue mass within a large central airway, without characteristic features to distinguish the mass from other tumors (Figure 4-59).
B 4-56 Squamous cell carcinoma of the trachea. Posteroanterior chest radiograph (A) and computed tomography scan of the distal trachea (B) reveal a round soft tissue mass arising from the posterior wall of the trachea. FIGURE
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4-57 Adenoid cystic carcinoma of the larynx and trachea. Anteroposterior (A) and lateral (B) tomograms of the larynx and proximal trachea and computed tomography scans at the level of the hyoid bone (C) and proximal trachea (D) reveal a smooth nodular tumor with endophytic growth. FIGURE
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FIGURE 4-58 Adenoid cystic carcinoma of the distal trachea and proximal left main bronchus. A, Anteroposterior tomogram of the trachea and carina reveals a smooth, well-defined mass arising from the left lateral wall of the trachea extending into the proximal left main bronchus. Computed tomography scans of the distal trachea (B, C) and proximal left main bronchi (D, E) demonstrate nodular thickening of the tracheal and left main bronchial walls. F, A postoperative anteroposterior tomogram of the trachea demonstrates a patent anastomosis following a carinal reconstruction.
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FIGURE 4-59
Mucoepidermoid carcinoma. Anteroposterior tomogram of the trachea (A) and computed tomography scan of the distal trachea (B) reveal a well-defined rounded mass in the distal trachea. A virtual bronchoscopic image (C) and the gross pathologic specimen (D) reveal an obstructing soft tissue mass in the distal trachea.
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CARCINOID TUMORS. Carcinoid tumors are neuroendocrine tumors derived from Kulchitsky’s cells.88 The typical carcinoid tumor represents the lowest grade subtype of a spectrum of tumors that includes the more aggressive atypical carcinoid tumor and the highly malignant small cell carcinoma. Typical carcinoids present in the fifth and sixth decades and tend to arise in the central bronchi, peripheral lung (10%), and rarely in the trachea. They tend to be smooth, well-defined, round masses that present as a nodular-filling defect, and may be associated with atelectasis, distal pneumonia, and/or bronchiectasis if they cause bronchial obstruction (Figure 4-60). Atypical carcinoid tumors tend to present in the sixth and seventh decades of life, may be either central or peripheral in the lung, and have a tendency to metastasize
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to regional hilar and mediastinal lymph nodes (Figure 4-61). Small cell carcinomas are extremely malignant tumors that present in the seventh and eighth decades. They are usually associated with large, bulky central hilar and mediastinal lymphadenopathy and distant metastases at the time of diagnosis. CT scans often reveal a small peripheral primary tumor within the lung, generally not visible on routine chest radiographs. Carcinoid tumors have several distinguishing features on imaging studies. Typical carcinoid tumors generally exhibit slow growth and may contain calcifications. Carcinoid tumors are highly vascular and will demonstrate a high degree of contrast enhancement with iodinated contrast on CT scans (Figure 4-62).
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FIGURE 4-60 Typical carcinoid tumor. Posteroanterior chest radiograph (A) and anteroposterior tomogram (B) reveal an obstructing mass in the left main bronchus with partial volume loss in the left lung. C, A contrast enhanced computed tomography scan demonstrates a focal mass within the left main bronchus (arrow). D, An octreotide scan of the chest demonstrates a focal area of intense uptake in the left hilum (arrow).
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FIGURE 4-61 Atypical carcinoid tumor. Posteroanterior chest radiograph demonstrates a peripheral mass in the right upper lobe, associated with right hilar adenopathy.
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FIGURE 4-62 Typical carcinoid tumor. A, Posteroanterior chest radiograph demonstrates a left hilar mass. B, Computed tomography scan with contrast enhancement reveals intense enhancement of the carcinoid tumor in the left lower lobe bronchus.
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Because somatostatin receptors are found in carcinoid tumors, radionuclide-coupled somatostatin analogues such as 123I-Tyr3-octreotide and 111In-octreotide can be used to identify carcinoid tumors. This diagnostic approach is helpful in identifying occult carcinoid tumors in those patients who present with clinical symptoms referable to serotonin, adrenocorticotropic hormone, or bradykinin production.
Mesenchymal Tumors Mesenchymal tumors are rarely reported to occur in the trachea, and tend to occur in young adults. Fibrosarcoma, leiomyosarcoma, chondrosarcoma, hemangioendotheliosarcoma, and lymphomas have been reported (Figure 4-63).89 Except for calcifications in chondrosarcomas, there are no specific characteristics with which to differentiate mesenchymal tumors from other malignancies (Figure 4-64).
Secondary Malignant Tumors Carcinomas, especially papillary and follicular types arising from the thyroid gland, may invade the larynx and cervical trachea in up to 5% of cases. The trachea may also be invaded by tumors of the esophagus and lung. The delineation of the extent of these tumors is best accomplished with CT and MRI (Figure 4-65).79,80
Tracheobronchomegaly Tracheobronchomegaly, seen in Mounier-Kuhn syndrome, is an abnormal diffuse dilatation of the trachea and main bronchi.90–92 The diagnosis of tracheobronchomegaly is usually apparent on chest radiographs, CT, or MRI. Typically, there is protrusion of the mucosa through the trachealis muscle between the cartilaginous rings, producing a scalloped or corrugated appearance of the trachea and main bronchi (Figure 4-66). There is often an abrupt transition between the dilated lobar bronchi and normal segmental bronchi. Patients with this disorder may have repeated respiratory infections leading to peripheral bronchiectasis.
B 4-63 Tracheal lymphoma. A, Anteroposterior tracheal tomogram reveals a smooth stenosis of the proximal trachea with an hourglass configuration. B, Computed tomography scan demonstrates thickening of the tracheal wall.
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FIGURE 4-64 Chondrosarcoma of the trachea. A, Posteroanterior chest radiograph demonstrates a large mediastinal mass that deviates and partially obstructs the mid and distal trachea. B, Computed tomography scan through the midtrachea reveals a large soft tissue mass containing calcification that encircles and narrows the trachea, consistent with a chondrosarcoma arising from the trachea.
B 4-65 Papillary carcinoma of the thyroid with laryngeal invasion. A, Lateral view of the neck shows a posterior supraglottic laryngealpharyngeal mass. B, Computed tomography section reveals a mass to the right and posterior to the larynx. There is destruction of the cricoid and thyroid cartilage on the right, with intraluminal tumor extension (asterisk). FIGURE
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Tracheobronchomegaly is also seen in association with Ehlers-Danlos syndrome, cutis laxa, and ataxia telangiectasia (immune deficiency syndrome), and with diffuse pulmonary fibrosis.
Tracheobronchomalacia Tracheobronchomalacia refers to a weakness in the tracheobronchial walls due to a primary immaturity or secondary softening or destruction of the cartilaginous rings, or excessive flaccidity of the posterior mem-
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FIGURE 4-66 Mounier-Kuhn syndrome. Anteroposterior (A) and lateral (B) tomograms of the trachea demonstrate dilatation of the trachea and main bronchi, with a scalloped or corrugated appearance. Computed tomography scans of the midtrachea (C) and main bronchi (D) demonstrate dilated scalloped-appearing airways.
branous wall with resultant collapsibility. Tracheobronchomalacia may be focal or diffuse.93,94 There are various causes of tracheobronchomalacia, including 1) congenital absence or hypoplasia of the cartilaginous rings, 2) traumatic causes related to mechanical ventilation with cuffed tubes or blunt chest trauma, 3) inflammatory conditions such as relapsing polychondritis, or 4) compression by vascular structures such as aberrant vessels or aneurysms or external mediastinal masses such as long-standing goiters. The location and extent of malacia is best evaluated radiographically by inspiratory/expiratory CT scans (Figure 4-67).
Acquired Tracheobronchoesophageal Fistulae Tracheobronchoesophageal fistulae may be congenital or acquired.95,96 The most common congenital tracheoesophageal fistula, accounting for 80 to 90% of cases, is esophageal atresia with a low tracheoesophageal
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4-67 Tracheobronchomalacia. Inspiratory (A) and expiratory (B) computed tomography scans of the main bronchi reveal marked collapse of the bronchi on expiration. There is also diffuse thickening and calcification of the airways, typical of relapsing polychondritis.
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A 4-68 Congenital “H-type” esophagobronchial fistula. A, Posteroanterior chest radiograph demonstrates right basilar pneumonia and right hilar adenopathy in a patient who experienced recurrent pneumonia in the right lung. B, A barium esophagogram demonstrates a small fistula extending from the distal esophagus to a right lower lobe bronchus. FIGURE
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FIGURE 4-69 Esophagobronchial fistula and broncholith due to histoplasmosis. An esophagogram (A) reveals an esophagobronchial fistula extending from the distal esophagus to the bronchus intermedius, and as demonstrated by a chest computed tomography scan (B), this patient had a broncholith (arrow) in the bronchus intermedius.
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fistula, presenting at birth. The “H-fistula” without atresia may be difficult to diagnose and may go undetected until adulthood (Figure 4-68). The majority of tracheobronchoesophageal fistulae in adults are acquired. Such acquired fistulae result from several causes, including 1) malignancy of the esophagus, trachea, bronchi, thyroid, and lymphomas; 2) radiation injury; 3) fungal infections such as histoplasmosis and actinomycosis, tuberculosis, syphilis, and bacterial infections (Figure 4-69); and 4) traumatic causes such as those from mechanical ventilation with cuffed tubes and indwelling nasogastric tubes, blunt or penetrating trauma, lye burns of the esophagus, instrumentation, or esophageal foreign bodies (Figure 4-70). The diagnosis of a fistula can be established with the judicious use of an esophagogram, using low osmolar contrast material. High osmolar contrast should be avoided, because if the lung becomes flooded with contrast, lifethreatening pulmonary edema and bronchospasm may develop.
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FIGURE 4-70 Postintubation tracheoesophageal fistula. Anteroposterior view of an esophagogram reveals a fistula of the proximal esophagus extending to the trachea in a patient with a tracheostomy tube.
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Lenique F, Brauner MW, Grenier P, et al. CT assessment of bronchi in sarcoidosis: endoscopic and pathologic correlations. Radiology 1995;194:419–32. Feldman F, Seaman WB, Baker DC. The roentgen manifestations of scleroma. Am J Roentgenol 1967; 101:807–813. Stern MG, Gamsu G, Webb WR, Stulbarg MS. Computed tomography of diffuse tracheal stenosis in Wegener granulomatosis. J Comput Assist Tomogr 1986; 10:868–70. Bhalla M, Grillo HC, McLoud TC, et al. Idiopathic laryngotracheal stenosis: radiographic findings. Am J Roentgenol 1993;1661:515–7. Mendelson DS, Som PM, Crane R, et al. Relapsing polychondritis studied by computed tomography. Radiology 1985;157:489–90. Im J-G, Chung JW, Han SK, et al. ST manifestations of tracheobronchial involvement in relapsing polychondritis. J Comput Assist Tomogr 1988;12:792–3. Onitsuka H, Hirose N, Watanake K. Computed tomography of tracheopathia osteoplastica. Am J Roentgenol 1983;140:268–70. Urban BA, Fishman EK, Goldman SM, et al. CT evaluation of amyloidosis: spectrum of disease. Radiographics 1991;13:1295–308. McCarthy MJ, Rosado-de-Christenson ML. Tumors of the trachea. J Thorac Imaging 1995;10:180–98. Caldarola VT, Harrison EG Jr, Clagett OT, et al. Benign tumors and tumorlike conditions of the trachea and bronchi. Ann Otol 1974;73:1042–61. Jones SR, Mers EN, Barnes L. Benign neoplasms of the larynx. Otolaryngol Clin North Am 1984;17:151–78. Aspestrand F, Kolbenstvedt A, Boysen M. CT findings in benign expansions of the larynx. J Comput Assist Tomogr 1989;13:222–5. Aywlard TD, Glege JB Jr. Primary papilloma of the trachea. Ann Thorac Surg 1973;16:620–3. Hyams VJ, Rabuzzi DD. Cartilaginous tumors of the larynx. Laryngoscope 1970;80:755–67. Sutton TJ, Nogrady MB. Radiologic diagnosis of subglottic hemangioma in infants. Pediatr Radiol 1973; 1:211–6. Cummings CW, Montgomery WW, Balogh K Jr. Neurogenic tumors of the larynx. Ann Otol Rhinol Laryngol 1969;78:76–95. Cotelingam JD, Barne L, Nixon VB. Pleomorphic adenoma of the epiglottis. Arch Otolaryngol 1977; 103:245–7. Som PM, Nagel BD, Feuerstein SS, Strauss L. Benign pleomorphic adenoma of the larynx. Ann Otol Rhinol Laryngol 1979;88:111–2. Oliveira CA, Roth JA, Adams GL. Oncolytic lesions of the larynx. Laryngoscope 1977;87:1718–25. Agarwal RK, Blitzer A, Perzin KH. Granular cell tumors of the larynx. Otolaryngol Head Neck Surg 1979;87:807–14. Schaefer SD, Blend BL, Denton JC. Laryngeal paragangliomas: evaluation and treatment. Am J Otolaryngol 1980;1:451–5. Pahor AL. Lipoma of the larynx. Ear Nose Throat J 1976;5:341–2. Johnson JT, Pouschter DL. Fibrous histiocytoma of the subglottic larynx. Ann Otol Rhinol Laryngol 1977; 86:243–6. Modlin B. Rhabdomyoma of the larynx. Laryngoscope 1982;92:580–2. Glazer HS, Mauro MA, Aronberg DJ, et al. Computed tomography of laryngoceles. Am J Roentgenol 1998;140:549–52. Silverman PM, Korobkin M. Computed tomographic evaluation of laryngoceles. Radiology 1982;145:104.
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Tanaka H, Mori Y, Kurokawa K, et al. Paratracheal air cysts communicating with the trachea: CT findings. J Thorac Imaging 1997;12:38–40. Sessions DG, Ogura JH, Fried MP. Carcinoma of the subglottic area. Laryngoscope 1975;85:1417. Pilch BZ, Dorfman DM, Brodsky GI, et al. Pathology of laryngeal malignancies. In: Fried MP, editor. The larynx: a multidisciplinary approach. 2nd ed. St. Louis: CV Mosby; 1996. Becker M, Moulin G, Kurt AM, et al. Non-squamous cell neoplasms of the larynx: radiologic-pathologic correlation. Radiographics 1998;18:1189–209. Takashima S, Takayama F, Wang Q, et al. Differentiated thyroid carcinomas. Prediction of tumor invasion with MR imaging. Acta Radiol 2000;41:377–83. Wang JC, Takashima S, Takayama F, et al. Tracheal invasion by thyroid carcinoma: prediction using MR imaging. Am Roentgenol 2001;177:929–36. Grillo HC, Suen HC, Mathisen DJ, Wain JC. Resectional management of thyroid carcinoma invading the airway. Ann Thorac Surg 1992;54:3–9. Hajdu SI, Huvos AG, Goodner JT, et al. Carcinoma of the trachea: clinicopathologic study of 41 cases. Cancer 1970;25:1448–56. Felson B. Neoplasms of the trachea and main stem bronchi. Semin Roentgenol 1983;18:23–37. Janower ML, Grillo HC, MacMillan AS Jr, et al. The radiologic appearance of carcinoma of the trachea. Radiology 1970;96:39–43. Cleveland RH, Nice CM Jr, Ziskind J. Primary adenoid cystic carcinoma (cylindroma) of the trachea. Radiology 1977;122:597–600.
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Stark P. Das adenoidzystische Karzinom (Zylindrom) der Trachea. Fortschr Rontgenstr (RoFo) 1982;136:31–5. Heitmiller RF, Mathisen DJ, Ferry JA, et al. Mucoepidermoid lung tumors. Ann Thorac Surg 1989; 47:394–9. Zwiebel BR, Austin JH, Grimes MM. Bronchial carcinoid tumors: assessment with CT of location and intratumoral calcification in 31 patients. Radiology 1991; 179:484–6. Weber AL, Shortsleeve M, Goodman M, et al. Cartilaginous tumors of the larynx and trachea. Radiol Clin North Am 1978;16:261–71. Dunne MG, Reiner B. CT features of tracheobronchomegaly. J Comput Assist Tomogr 1988;12:388–91. Katz I, LeVine M, Herman P. Tracheobronchomegaly: the Mounier-Kuhn syndrome. Am J Roentgenol Radium Ther Nucl Med 1962;88:1084–94. Shin MS, Jackson RM, Ho K-J. Tracheobronchomegaly (Mounier-Kuhn Syndrome); CT diagnosis. Am J Roentgenol 1988;150:777–9. Feist JH, Johnson TH, Wilson RJ. Acquired tracheomalacia: etiology and differential diagnosis. Chest 1975; 68:340. Aquino S, Shepard JO, Ginns LC, et al. Acquired tracheal malacia: detection by expiratory CT scan. J Comput Assist Tomogr 2001;25:394–9. Judd DR, Dubuque T Jr. Acquired benign esophagotracheobronchial fistula. Dis Chest 1968;54:237–40. Berkmen YM, Young HA. CT diagnosis of acquired tracheoesophageal fistula in adults. J Comput Assist Tomogr 1985;9:302–4.
CHAPTER FIVE
Diagnostic Endoscopy Hermes C. Grillo, MD
General Considerations Techniques
General Considerations Laryngoscopy and bronchoscopy are always required to assess airway lesions. Esophagoscopy is added if the lesion is a tumor that may involve the esophagus, if there is a question of fistula, or if there is another reason to suspect esophageal pathology. Mediastinoscopy is not often done as a separate procedure for the assessment of primary tracheal lesions. Photographs of some lesions, as observed through bronchoscopy, are included in the color plates.
Laryngoscopy This procedure is performed to determine if there is pathology in the larynx, either as an extension of or in addition to tracheal pathology, and also to assess glottic function and competence (see Chapter 35, “Laryngologic Problems Related to Tracheal Surgery”). Functional evaluation of the larynx is made by indirect examination, or, now more commonly, with a flexible laryngoscope or bronchoscope. The conscious patient can cooperate in maneuvers necessary to show whether the vocal cords move normally. Glottic function may be assessed under general anesthesia by lightening the anesthesia to a point where vocal cord reflexes return, but this is not nearly as satisfactory. Mass movement may be difficult to interpret. Examination must be accurate and is often best performed by a consulting otolaryngologist. If any complexity is suspected, I usually arrange for an endoscopy as a joint effort with the otolaryngologist. Degrees of aspiration on deglutition are assessed by barium studies, conventional or modified. If patients have pathology in both the larynx and trachea, it is critically important to define both lesions at the outset, in order to plan effective treatment. Generally, a significant non-neoplastic laryngeal lesion should be corrected or made manageable by an initial laryngeal procedure before the tracheal lesion is surgically treated. Alternatively, both laryngeal and tracheal lesions may be repaired concurrently, although this usually requires concomitant tracheostomy for safety, which is not always desirable because the surgeon might correct a tracheal lesion by extensive resection, possibly bringing the larynx down to the sternal notch. If an obstructive lesion in the larynx is only then detected, tracheostomy may be difficult or
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dangerous to the fresh anastomosis, either to the larynx itself or to an adjacent brachiocephalic artery. Intubation, preferably avoided following fresh tracheal reconstruction, could be necessary. Laryngoscopic examination is particularly important in patients with trauma to the upper airway, where recurrent laryngeal nerve injuries are likely. Many patients with postintubation tracheal stenosis have inflammatory lesions of the larynx as well, which may cause obstruction in concert or independently. Vocal cord paralysis may also be present in these patients with or without a history of prior tracheal reconstruction or of direct injury to the larynx. Cord paralysis also occurs with primary or secondary tracheal neoplasms, and high tumors may invade the larynx.
Bronchoscopy For a tracheal lesion, bronchoscopy has two purposes. The first is to obtain precise diagnosis of the type and extent of the lesion. The second is to treat obstruction, if present, in order to provide an airway adequate for immediate resection, to allow delay for further studies or treatment prior to resection, or to permit safe nonsurgical treatment such as stenting or irradiation (see Chapter 40, “Tracheal and Bronchial Stenting” and Chapter 41, “Radiation Therapy in the Management of Tracheal Cancer”). For the first purpose, I prefer rigid bronchoscopy. For therapeutic purposes, the rigid instrument is essential. Hopkins telescopes used through the rigid bronchoscope provide a magnified image, which is very much clearer optically than that provided even with improved flexible fiber optics. This provides precise delineation of the extent of glottic and subglottic injury and of the tracheal lesion. Measurements of distances between major normal and pathologic landmarks are more accurately made with the rigid bronchoscope. Furthermore, the rigid bronchoscope affords better control of the airway while these observations are being made. The flexible bronchoscope is useful for visualization and for diagnostic maneuvers such as biopsy, brushing, and aspiration. It is especially useful for precise positioning of endotracheal tubes, guiding catheters or stents for intraoperative localization of lesions, and for examination of a new anastomosis. Usually, radiologic examination of the airway precedes bronchoscopy. These images serve as a road map for the bronchoscopist. Obviously, there are cases of suspected airway obstruction where bronchoscopy is done initially and sometimes urgently. If significant obstruction is identified, it is preferable not to instrument the lesion unless urgently required to, in order to avoid precipitating acute obstruction, unless the operator is prepared to proceed with whatever is necessary to relieve the obstruction, either by endoscopy or surgically. Definitive endoscopic examination is best accomplished under general anesthesia with rigid instruments, and most often immediately before a surgical operation to correct the lesion. This avoids any hazard of precipitating obstruction during diagnostic examination and avoids a second anesthesia. Definitive histologic diagnosis may be obtained prior to resection by dependable frozen sections from the generous biopsies permitted via rigid bronchoscopy. A limited lesion that clearly needs resection may not require preliminary biopsy. Control of post-biopsy bleeding under general anesthesia with a rigid bronchoscope in place has, in our hands, never presented a major problem. If this were to occur, tamponade with a cuffed endotracheal tube should provide control, followed if need be by surgery. Lesions that appear excessively vascular on telescopic examination should best not be biopsied. Aneurysmal vessels or hemangiomatous appearance are indications for angiography. Bronchoscopy, as a separate procedure prior to surgery, is justifiable where the lesion is particularly complex and requires special planning, where reparative or extirpative surgery seems highly unlikely, if the patient will require a prolonged period of preparation for surgery, or if frozen sections are unlikely to define the type or extent of an unusual tumor. Virtual bronchoscopy using three-dimensional reconstruction from helical computed tomography images offers a noninvasive technique for initial evaluation of tracheal or bronchial lesions, but it does not replace the refinement and precision of an actual bronchoscopy.1
Diagnostic Endoscopy
Therapeutic bronchoscopy for obstruction can be a critical procedure and is discussed in Chapter 19, “Urgent Treatment of Tracheal Obstruction.” I do not believe a surgeon should attempt tracheal reconstructions unless he is competent in and equipped to perform rigid bronchoscopy.
Esophagoscopy This technique is performed when any preceding imaging studies, usually with relation to a tumor, indicate deformation or possible involvement of the esophagus. Often, tumors deform the esophageal wall without actually extending into or through the mucosa. Nonetheless, it is wise to add esophagoscopy, following bronchoscopy. Esophagoscopy may be useful to visualize tracheoesophageal fistulae after first examining the defect bronchoscopically. In the rare patient with a concomitant esophageal stricture at the level of a tracheal stricture, or with an unrelated distal stricture, these lesions should be evaluated, and if necessary, dilated.
Mediastinoscopy This procedure has shown little value for evaluation of primary tracheal tumors, particularly adenoid cystic carcinoma. The finding of involved lymph nodes adjacent to the tumor on one or both sides of the trachea does not make a patient unsuitable for surgical resection. These paratracheal lymph nodes are the first regional lymph node stations involved by a tracheal tumor and do not seem to carry the same significance for adenoid cystic carcinoma of the trachea as they do for carcinoma of the lung. I consider these to be N1 nodes with respect to a tracheal tumor. If the primary tracheal tumor is large, and invasion of other mediastinal structures is suspected, then mediastinoscopy may be performed immediately prior to a planned exploration for resection under the same anesthesia. Surgical planes will thus not be confused by inflammatory scarring, which results if resection is delayed following mediastinoscopy. Histologic information from mediastinoscopy is obtained by frozen section. This permits surgeons with final responsibility for tracheal or carinal resection and reconstruction to make their own decisions, and to operate in a freshly dissected field, where iatrogenic changes do not confuse basic pathology. Mediastinoscopy does not provide any better mobilization of the trachea than can be obtained by retrograde pretracheal dissection at thoracotomy. In evaluating bronchogenic carcinomas that invade the main bronchus or carina, it is also preferable to defer the necessary mediastinoscopy to the time of a potential carinal resection. If N2 lymph node involvement is discovered, which might dictate delay for a program of adjunctive preoperative treatment, consequent inflammatory changes must be accepted.
Techniques This chapter assumes familiarity and competence with the endoscopic techniques and instruments discussed herein, including facile use of the rigid bronchoscope.
Laryngoscopy If it is to be performed concurrently with rigid bronchoscopy, laryngoscopy is done first, almost always under general anesthesia. Where laryngeal complexities are suspected or where it is possible that a laryngeal surgical procedure will have to be done at the time of the examination, or later as an independent but preceding procedure, a consulting otolaryngologist should be present. Often, the consulting otolaryngologist will have examined the patient previously, and already have performed an indirect or flexible laryngoscopy. We find the Holinger anterior commissure laryngoscope to be particularly useful for examining the glottis and subglottic larynx under anesthesia (Figure 5-1A). The Lewy suspension apparatus is frequently useful (Figure 5-1B). Anesthesia or ventilation may be easily maintained by intermittent placement of an endotracheal tube through the laryngoscope. Hopkins optical telescopes provide a clear, magnified view
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FIGURE 5-1 A, A Holinger anterior commissure laryngoscope, especially useful for direct visualization of the glottis and subglottic larynx. B, The Lewy suspension apparatus in use. The angle of the laryngoscope is fully adjustable.
A
B
through the laryngoscope. Minor laryngeal procedures, such as removal of vocal cord polyps or granulomas, or even division of a posterior commissural stricture between the arytenoids, may be accomplished at this time. If considerable manipulation is required, it is preferable not to proceed at once to a further tracheal procedure, since glottic edema may become a problem postoperatively. In this way, tracheostomy may be avoided. If the laryngeal lesion is of great complexity, such as a complete subglottic stenosis that will require laryngofissure and stenting, the patient may be transferred to the care of the otolaryngologist until that problem is resolved. Endoscopic examination may reveal details that were not evident from radiologic images, particularly in a high subglottic laryngotracheal lesion. I prefer to be certain that a complex laryngeal reconstruction has truly succeeded before proceeding to an extended tracheal or laryngotracheal reconstruction. Such patients often already have a distal tracheostomy in place, which is removed at the time of the subsequent reconstruction.2 Maddaus and colleagues preferred to perform repair of both lesions concurrently, establishing a prolonged tracheostomy with stent placement for later removal.3
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A B C
D
FIGURE 5-2
Basic equipment for a rigid bronchoscopy. From top to bottom: A, Jackson bronchoscope (Pilling) with a ventilatory sidearm and eyepiece cap at the right. Bronchoscopes of internal diameter 7, 8, and 9 mm are routinely provided for adult examination. B, Metal centimeter ruler. C, Storz Hopkins telescope with a “gasket” to seal the bronchoscope. Telescopes of 0˚, 30˚, and 90˚ are made available in the kit. D, Adequate large-bore suction tip, especially useful for brisk or massive hemoptysis, thick secretions, biopsy, or coring-out procedures.
Rigid Bronchoscopy This technique best entails general anesthesia, which may be given safely even in the face of severe airway obstruction (see Chapter 18, “Anesthesia for Tracheal Surgery”). Numerous techniques have been described for the anesthetic management of rigid bronchoscopy under general anesthesia, including jet ventilation. I have found the Jackson ventilating bronchoscope, with a side port for attachment of an anesthesia tube and window eyepiece or telescopic gasket, to be highly satisfactory. The surgeon must be on hand during induction of anesthesia, especially when there is any degree of airway obstruction. Equipment for relief of obstruction must be immediately available if obstruction is present, and the anesthetist must have confidence that the surgeon can establish an airway at any moment. This presupposes complete competence in rigid bronchoscopy, which unfortunately seems in danger of becoming a lost art. Our bronchoscopic set-up for examination of an adult includes 7, 8, and 9 mm rigid Jackson ventilating bronchoscopes (Figure 5-2). The tips of these bronchoscopes are oblique with rounded edges, and are easier to introduce through a tight stenosis or past a tumor than are the spade-like tips of the Storz instruments, which may also elevate a flap of mucosa (Figure 5-3). A large bore suction tip must be available. The Storz Hopkins telescopes are introduced through adapters placed over the open end of the rigid bronchoscope to provide a seal and permit ventilation (see Figure 5-2). When the telescope is not in place, a window cap is placed over the proximal end of the bronchoscope. Also available in the endoscopic room or immediately adjacent should be a kit of sterile pediatric Jackson rigid bronchoscopes of the following sizes: 3.5, 4, 5, and 6 mm. These may be used serially to dilate tightly stenotic lesions, as described in Chapter 19, “Urgent Treatment of Tracheal Obstruction.” The Storz pediatric bronchoscopes are superb for diagnosis in children, but for dilation, the tip of the Jackson pediatric bronchoscope is preferable. If it is known that a high degree of obstruction is present, these bronchoscopes should be on the instrument table initially, along with selected small bougies (see Figure 19-1 in Chapter 19, “Urgent Treatment of Tracheal Obstruction”). Even if the patient has a preexisting tracheostomy, bronchoscopic examination is commenced through the mouth, passing the rigid bronchoscope into the larynx and through the glottis as far distally as
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FIGURE 5-3
Comparative tips of Jackson (above) and Storz (below) rigid bronchoscopes. The Jackson model is more easily introduced into a tightly stenotic lesion with a rotatory motion than the Storz model, with its more shovel-like tip.
is easily permitted. This provides a picture of the anatomy of the entire airway and more precise measurements of lengths of lesions and normal structures. If the obstructing lesion is not densely fibrotic, or if it is a tumor, then the bronchoscope is passed beyond the lesion. If the lesion is densely stenotic, particularly if it involves the subglottic larynx, or if there is discontinuity between the proximal airway and the more distal trachea, as occurs sometimes with postintubation stenoses or after trauma, then maximum information is obtained by examining the airway up to the point of obstruction, followed by completion of the examination of the distal trachea through the tracheostomy present. Once the bronchoscope is passed and the airway is cleared of secretions (which may pool distal to a partially obstructing lesion), and also cleared of blood that may be incited by the examination, the 0˚ telescope is used first for visualization. Desired information about the larynx includes 1) status of both the structure and function of the glottis, including vocal cords, arytenoids, and the anterior and posterior commissures; and 2) airway diameter, deformities, stenosis, inflammation, edema, or involvement by tumor of the subglottic larynx down to the inferior margin of the cricoid cartilage. The cricoid cartilage may be directly visualized, if not altered by pathology. It presents as a broad ring immediately above the narrower tracheal rings (Figure 5-4). If pathology has blurred this differential, gentle external pressure on the palpable cricoid cartilage by the bronchoscopist’s finger, while looking through the 0˚ telescope, usually defines the exact level. For greatest precision, a no. 25 hypodermic needle may be passed through the skin and wall of the trachea or larynx, just below the cricoid, and its entry point into the airway noted. In adult males of average size, an 8 or 9 mm bronchoscope (40 cm long) is used initially. In smaller males or in females, the 7 mm bronchoscope is used initially. For visualization and manipulation of upper tracheal lesions (such as injection of triamcinolone or Depo-Medrol into lesions with special long hypodermic needles), a 9 mm bronchoscope, which is only 25 cm long, is useful (Figure 5-5). After the larynx has been fully examined, the bronchoscope is passed distally, observing the tracheal configuration in anteroposterior and lateral directions and for abnormalities in the cartilaginous architecture. The membranous wall is characteristically smooth. The mucosa is examined for inflammation, easy bleeding or increased vascularity, extrinsic or submucosal deformities, and mucosal lesions. Areas of malacia can sometimes be detected, assisted by gentle palpation in the neck or by observing collapse with respiration as the patient’s anesthesia is lightened. Malacia is better identified with the flexible bronchoscope
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5-4 Endoscopic delineation of the border between the inferior margin of cricoid cartilage and the uppermost tracheal rings. The cricoid is very broad compared to the narrow tracheal rings below. Bronchoscopic measurements of the tracheal length accurately use this point proximally. However, if the cricoid is indistinct or obscured by pathologic change, the glottis is used as the proximal landmark, but the approximate length of the subglottic larynx (to bottom of cricoid) must be subtracted to obtain the tracheal length.
FIGURE
FIGURE 5-5 Equipment for intralaryngeal or intratracheal injection. A no. 20 needle with no. 25 tip is long enough to reach through an adult rigid bronchoscope (8 mm, 40 cm shown). A tuberculin syringe is necessary to develop sufficient pressure to inject fluid via the long needle with this fine tip. Also shown is a short bronchoscope (9 mm, 25 cm) to facilitate access to laryngeal and subglottic areas.
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under topical anesthesia so that the patient may cooperate in maneuvers that emphasize collapse, usually cervical inspiratory collapse or intrathoracic expiratory collapse. Complete bronchoscopic examination to segmental levels is particularly important in patients with squamous cell cancer of the trachea, since they may well have concurrent squamous cell lesions of the aerodigestive tract. A flexible bronchoscope is easily passed through the telescopic “adapter” via the rigid bronchoscope to complete a distal survey of segmental bronchi. These patients also deserve complete endoscopic examination of the pharynx and esophagus, in addition to laryngoscopy and bronchoscopy. The endoscopist should be familiar with normal and abnormal variations in the trachea (see Figure 1-2 in Chapter 1, “Anatomy of the Trachea”). Sabre-sheath trachea has been mistakenly identified as stenosis (see Chapter 14, “Infectious, Inflammatory, Infiltrative, Idiopathic, and Miscellaneous Tracheal Lesions”). Tracheopathia osteoplastica is very rare and may not be recognized when observed (see Chapter 15, “Tracheobronchial Malacia and Compression”). Findings in children and in congenital lesions of the trachea are described in Chapter 6, “Congenital and Acquired Tracheal Lesions in Children.” Even the most severe deviations of the trachea due to extrinsic pressure, as seen with huge substernal and intrathoracic goiters, usually allow easy passage of a rigid bronchoscope (see Chapter 15, “Tracheobronchial Malacia and Compression”). The trachea widens and straightens out as the rigid bronchoscope is passed. Normal contraction of the posterior membranous wall, and its protrusion forward in response to cough, either in the awake patient or under lightened anesthesia, should not be confused with malacia. Pulsatile compression should especially be noted. Pulsation and extrinsic deformation is commonly seen in the left lateral wall of the lower trachea due to the adjacent aorta. Bronchoscopes should not be pushed through a fibrous stenotic lesion with excessive force. Dilation should be carried out systematically (see Chapter 19, “Urgent Treatment of Tracheal Obstruction”) to avoid splitting or perforating the tracheal wall proximal to the tough fibrous stricture of a postintubation stenosis. In the case of tumors, one can always initially pass the rigid bronchoscope beside the tumor, on the side where tumor is not attached to the tracheal wall, no matter how complete the obstruction appears to be. Cartilage will yield and permit the bronchoscope to pass, partly displacing the tumor. The bronchoscope can be passed through the center of the circumferential tumor, carefully following even a tiny opening. A small bougie can serve as a guide. Cardiopulmonary bypass has been unnecessarily employed to resect tumors that appear to be causing nearly complete obstruction. A severely obstructing tumor can be cored out using the rigid bronchoscopes, with additional trimming done with biopsy forceps (see Chapter 19, “Urgent Treatment of Tracheal Obstruction”). The technique is so simple that we have found it unnecessary to use the laser for this purpose.4 Bleeding has not been a problem, despite cautions and alarms cited as rationale for laser removal of obstructing tumor. I routinely measure findings, including the location of normal structures and of the upper and lower limits of lesions. This is performed by difference, as illustrated in Figure 5-6. Systematic measurement is begun with the tip of the bronchoscope touching the carina, as seen through the 0˚ telescope, which is kept just proximal to the tip of the bronchoscope. Measurement is made of the distance from a selected point on the upper teeth or gingival ridge to a fixed point at the hub of the bronchoscope, using a centimeter rule. The bronchoscope is then withdrawn until its tip is at the lower border of the lesion. Measurement is again made at this point and is repeated with the bronchoscope’s tip at the upper border of the lesion. Other points also noted are the location of a tracheal stoma, the level of the inferior margin of the cricoid cartilage, and the level of the vocal cords. Distances are obtained by subtraction from the figures recorded (Figure 5-7). Even with the most meticulous measurement, such figures are accurate only to about 0.5 cm because of the imprecise level of points of measurement and the flexibility of tissues. I find it helpful to construct a diagram of the airway and its lesions (see Figure 5-7F). It is posted in the operating room for reference and a copy is recorded in the patient’s chart.
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A
B
C
D
E
FIGURE 5-6 Localization and measurement of the length of components of the upper airway and of a lesion. A telescope is just within the bronchoscope to locate the tip of the bronchoscope precisely. A, With the tip of the bronchoscope at the carinal spur, distance is measured and recorded between the incisor teeth (or upper gingival ridge) and a proximal fixed point on the bronchoscope. Similar measurements are made with the bronchoscopic tip successively at the lower edge of the lesion (B), the upper edge of the lesion (C), the lower edge of the cricoid (D), and/or at the vocal cords. E, Recording of measurements and approximate lengths (by subtraction).
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B
C
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E 5-7 Quantitative recording of tracheal pathology. A, Tomographic cut of a trachea with the tracheostomy tube in place, stenosis and granuloma below (between arrows), a widened tracheal segment below this, and a short distal normal segment above the carina. This is a road map for the bronchoscopist. Bronchoscopic views correspond. View at: B, glottis (arrow) to site of stoma; C, stoma with tube in place; D, stenosis distal to stoma; and E, malacic and dilated trachea below stenosis with carina distantly seen. F, Diagram sketched in the operating room, from bronchoscopic measurements, using a marking pen on glove paper. Noted are the distances between vocal cords and tracheostome (dotted lines; 1 cm), stoma and stenosis (1.5 cm), length of stenosis (2.5 cm), and distal trachea from stenosis to carina (4 cm). A granuloma is roughly outlined above the stenosis, and stippling distal to the stenosis locates malacia above a few remaining, relatively normal tracheal rings. The outlines at the left represent configuration of this severely damaged trachea at levels where they were observed. Total tracheal length is about 8 cm. FIGURE
F
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During or following extensive rigid laryngoscopy and bronchoscopy, especially if manipulation has been necessary, Decadron is administered for 24 to 48 hours, beginning intraoperatively, in an effort to minimize edema. Racemic epinephrine is also often added.
Flexible Bronchoscopy The flexible bronchoscope does not replace the rigid instrument in diagnosis nor in management of airway lesions, but it is a very useful adjunctive tool. It should be used liberally by pulmonologists to rule out organic obstruction in patients thought to have “adult onset asthma,” to clarify the origin of hemoptysis (however minor), and to investigate the possible causes of recurrent or unyielding volume loss, atelectasis, or pneumonitis. Intubation, for any reason, is facilitated by using the flexible bronchoscope as a guide. Difficult intubations are made simple in this way. Traumatic tracheal separation may respond to this technique. Rigid bronchoscopy is truly impossible in only very few patients. Examples included 2 patients with achondroplastic dwarfism with prognathous jaws, which produced a deep right angle between the oropharynx and the trachea. Another patient suffered severe fixed cervical deformity as a result of radical neck surgery and remote high-dose irradiation. In such patients, only flexible bronchoscopy is possible. If general anesthesia is elected, intubation may be avoided by passing the flexible bronchoscope through a laryngeal mask airway (Figure 5-8A).5 The flexible instrument may be used with the sealing cap used for telescopes through a rigid bronchoscope, in order to expand examination to include segmental bronchi. It is also introduced via an adapter through an endotracheal tube to identify a precise point in the airway during surgery (Figure 5-8B). If necessary, the endotracheal tube is partly withdrawn and the bronchoscopic light transilluminates the trachea to the operative field (with operating table lights deflected). Thus, the extremities of a postintubation stenosis, not clearly defined visually from the outside of the trachea, can be identified precisely. The surgeon passes a no. 25 needle through the tracheal wall into the lumen, and the needle is adjusted precisely by bronchoscopic confirmation (see Figure 24-10 in Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection”). The flexible bronchoscope is also invaluable in placing and replacing either double or single lumen tubes in the left or right main bronchus during bronchial or carinal resections. A pediatric flexible bronchoscope can be passed through one of the channels of a double lumen tube. Tracheobronchial anastomosis can be examined intraoperatively in this way. Correction of postpneumonectomy syndrome, of splinting procedures for tracheobronchomalacia, and assessment for malacia after excision of huge compressive goiters are established by repeated intraoperative flexible endoscopy. Transillumination may help to identify the location of a bronchial stump, in the event of a chronic bronchopleural fistula being buried in dense, irradiated mediastinal cicatrix. Flexible bronchoscopy has become our routine method for examination of an anastomosis, prior to discharge of a patient recovering from tracheal reconstruction. It is much more dependable than any other imaging technique for the early identification of anastomotic defects of any type.
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A FIGURE 5-8 A, Laryngeal mask airway (LMA). A flexible bronchoscope is easily passed through the LMA. B, Adapter for bronchoscopy via endotracheal tube or LMA (Portex).
B
References 1. 2. 3.
Finkelstein SE, Summers RM, Nguyen DM, et al. Virtual bronchoscopy for evaluation of malignant tumors of the thorax. J Thorac Cardiovasc Surg 2002;123:967–72. Grillo HC, Mathisen DJ, Wain JC. Laryngotracheal resection and reconstruction for subglottic stenosis. Ann Thorac Surg 1992;53:54–63. Maddaus MA, Toth JLR, Gullane PJ, Pearson FG. Subglot-
4. 5.
tic tracheal resection and synchronous laryngeal reconstruction. J Thorac Cardiovasc Surg 1992;104:1443–50. Mathisen DJ, Grillo HC. Endoscopic relief of malignant airway obstruction. Ann Thorac Surg 1989;48: 469–75. Roberts JT. Clinical management of the airway. Philadelphia: WB Saunders; 1994. p. 365.
CHAPTER SIX
Congenital and Acquired Tracheal Lesions in Children Hermes C. Grillo, MD
Embryology of the Trachea Congenital Lesions Acquired Lesions Assessment Treatment and Results
Congenital tracheal lesions are rare, and acquired tracheal lesions in children are also relatively uncommon. Experience in their management is consequently quite limited and widely dispersed. The small diameter of the juvenile trachea can easily become obstructed following surgery. One millimeter of subglottic swelling can reduce a newborn airway to one-third of its normal cross-sectional area. The danger of separation or stenosis is increased after reconstruction, because the delicate structure of the tracheal wall tolerates anastomotic tension less well. The great length of many congenital lesions prohibits resection and presents special problems in correction. Earlier concerns about tracheal growth following anastomosis have been allayed by experimental and clinical observations.
Embryology of the Trachea The laryngotracheal groove or sulcus appears in the proximal foregut at the third week (3 mm embryo, stage 10).1 The laryngotracheal groove progresses caudad and the lateral ridges progress cephalad to form the primordium of the trachea (Figure 6-1). The pulmonary primordium appears and bulges ventrally from the foregut. Complete separation of trachea and esophagus occurs by 11 to 14 mm (sixth week). The tip of the tracheal primordium buds asymmetrically, left and right, at the 4 mm stage, to provide bronchial primordia. Mesenchymal proliferation by cells lining the coelomic cavity provides the tissue from which cartilage, muscle, and connective tissue will develop. Epithelial–mesenchymal interrelationships are essential for bronchial and pulmonary development to occur. The tracheal bifurcation moves gradually downward from the neck to the level of the fourth vertebra. Cartilage appears in the trachea at 10 weeks. When the laryngotracheal groove appears, the forerunner of the glottis also appears as a median slit in the pharyngeal floor between the fourth and sixth branchial arches. The epiglottic primordium lies anteriorly and the arytenoid swellings lie laterally prior to their more medial migration. Ventricular buds are solid at first. A T-shaped slit appears, which opens into a lumen by the eighth week. Vocal cords are seen at 3 months. Thyroid and cricoid cartilages appear between 5 and 7 weeks. The laryngeal cartilages derive from the fourth and fifth arches.
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FIGURE 6-1
Development of larynx and trachea at about the sixth to eighth weeks. The laryngeal slit lies in the floor of the pharynx. Numerals indicate arches. The foregut is separating into the trachea and esophagus. Stem bronchi and branches are present. Specific stages are altered to show general relationships. Adapted from Gray SW and Skandalakis JE.1
Failure of complete separation of the foregut into respiratory and alimentary components is the most common defect and produces tracheoesophageal fistula (TEF). At the upper end, the larynx may fail to reopen, producing atresia (a fatal anomaly), or it may fail to form a complete posterior septum, producing a laryngotracheoesophageal cleft. Tracheal atresia, stenosis, esophageal atresia, and tracheoesophageal fistula occur more distally. The relatively separate processes of laryngeal development and budding of bronchi and pulmonary development allow for malformations of the trachea, such as agenesis and stenosis in the presence of a normal larynx and bronchial tree.
Congenital Lesions Tracheal agenesis or atresia is usually fatal at birth. The larynx may form normally. The lungs may or may not be normal, and with or without bronchial communications to the esophagus (Figure 6-2). These malformations are extremely rare. The most common presentation is with normal bronchi, communicating centrally to the esophagus.2 Other congenital anomalies are common in these patients. Fonkalsrud and colleagues described a newborn of type C, who survived for a short term by using the esophagus as an airway.3 A major bronchus may also communicate directly with the esophagus while the balance of the lung is served by anomalous bronchi from a partly stenotic trachea. Microgastria is a common concomitant feature. No systematic surgical treatment has evolved, doubtlessly due to the rarity and variations in the anomalies as well as the complexity of the defects. Hiyama and colleagues described two such patients in whom diagnosis was suspected due to respiratory distress without audible cry and difficulty in intubation.4 One infant was successfully treated by the
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Tracheal agenesis, redrawn according to Faro and colleague’s classification.2 In addition to types diagrammed, type A (8%) shows total pulmonary agenesis and type G (5%) has short segment tracheal agenesis. In type B, both main bronchi connect to esophagus separately, whereas in type C, bronchi are fused but a bronchoesophageal fistula (BEF) is present. Type D additionally has an atretic band (dashed line) from larynx to BEF. Type E has a tracheoesophageal communication, but in type F, communication with the esophagus is absent. Distribution of types in Faro and colleague’s collected series of 39 patients is noted. FIGURE 6-2
following procedures: gastrostomy and abdominal esophageal banding, translaryngeal and esophageal ventilation by endotracheal tube, tracheostomy and later T tube, pharyngeal sump drainage followed by establishment of cervical esophagostomy (proximal tracheal segment present), and esophageal reconstruction by colonic interposition at age 3. In the more common anomaly of tracheoesophageal fistula, the tracheal problem is usually managed by division and closure of the communication. Congenital TEF and the complexities of esophageal atresia have been well described and categorized. The reconstructive challenge is principally esophageal, and congenital TEF is not further detailed here. Rarely, there is accompanying tracheal stenosis. Adjacent local tracheomalacia, especially after repair of TEF, may cause respiratory problems, and is described later. Less commonly found is an H-type congenital tracheoesophageal fistula without concomitant esophageal atresia, often high in the trachea and usually small in diameter. It is sometimes discovered in the adult and is usually managed by transcervical division and closure (Figure 6-3).5,6 Coughing is the usual symptom, especially after ingestion of liquids. A large fistula may be treated by limited tracheal resection and anastomosis, with esophageal closure. Congenital fistula between biliary and respiratory tracts is extraordinarily rare.7 Respiratory problems begin with cough and progress to intractable pneumonia. The most common location of the fistula is at the carina, but right and left main bronchial connections have been noted. Yellow fluid is identified bronchoscopically. Contrast will identify a long paraesophageal tract connecting to a hepatic duct. It has also been seen in a young adult.8 Excision of the intrathoracic segment with closure at the carina (or bronchus) and at the diaphragmatic level cures the problem. Congenital bronchoesophageal fistula is a rare anomaly with fewer that 150 cases reported (see Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula”).9 Symptoms may not occur until
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FIGURE 6-3 H-type tracheoesophageal fistula in a 49-year-old woman. A, Fistula demonstrated (arrow) by barium contrast swallow. The trachea is clearly outlined. B, Computed tomography scan showing the small fistula. The fistula was divided and repaired easily through a low collar incision. Also, see Figure 6-21B. (Courtesy of Dr. Hon Chi Suen.)
A
B
later in life (reported from age 9 to 83 years) but 25% of cases present before age 17. These fistulas connect to the middle or lower esophagus, from right upper lobe, from left lower lobe, from bronchus intermedius, from right middle or lower lobe, and from left upper lobe. The fistula usually slopes downward from the bronchus to esophagus, perhaps accounting in part for the lack of earlier symptoms, but it may also connect from a small diverticulum of esophagus, or on the pulmonary side, to a cyst or to a sequestrated lobe.10 (See Figure 12-10 in Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula.”) Cough, especially after drinking, is the most common symptom, with respiratory infection and hemoptysis, and even hematemesis (seen less often). Barium esophagogram is the most accurate method of diagnosis. Endoscopy or bronchography are less helpful. Methylene blue in the esophagus aids bronchoscopic identification. The condition is permanently corrected by surgical excision and closure of the fistula, with interposition of healthy tissue between the bronchus and esophagus. Chronic pulmonary infection may dictate limited resection of the lung (see Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula” and Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula”). A very rare anomaly is the laryngotracheoesophageal cleft. Varying degrees of incompleteness of the wall between the larynx and trachea and the esophagus present (Figure 6-4).11,12 A minor interarytenoid cleft reaching through cricoid cartilage, a deeper defect reaching to the upper trachea, and a complete cleft extending to the carina have been designated as types I, II, and III, respectively.11 Ryan and colleagues added type IV, where the defect extends into both main bronchi.12 Type I may not require repair. Type II has been repaired through laryngofissure or cervical or cervicomediastinal approach. Types III and IV are best approached laterally transcervically and transthoracically (see Chapter 33B, “ Repair of Congenital Tracheal Lesions: Larynogotracheoesophageal Cleft Repair”). Donahoe and Gee first successfully repaired a type III cleft, with long-term survival.13
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FIGURE 6-4 Laryngotracheoesophageal cleft of increasing severity. Posterior view, with esophageal wall opened to show extent of cleft. Based on Petterson’s classification.11 Adapted from Ryan DP et al.12
Tracheal webs sometimes occur in the neonatal and juvenile trachea at the cricoid level. Laryngeal webs at the glottic level are more common. The tracheal web does not involve any significant length of airway and is consequently usually treated endoscopically (Figure 6-5). Congenital cartilaginous cricoid stenosis is an extremely rare lesion.14 Subglottic stenosis is more common as a consequence of postintubation injury. Other purely laryngeal congenital lesions, such as glottic and subglottic stenosis and atresia, are not considered here. Segmental or diaphragm-like congenital main bronchial stenosis, principally on the right, has been very rarely encountered.15 Segmental bronchial resection or even wedge resection in the instance of a thin web is an effective treatment. In the latter case, bronchoscopic management might be considered. Congenital tracheal stenosis has been classified into three principal types: 1) generalized hypoplasia, 2) funnel-like narrowing, and 3) segmental stenosis (Figure 6-6).16 The stenotic segment is most often composed of completely circular “O” rings of cartilage (Figure 6-7). Alternatively, disorganized cartilages, ridges, or plates of cartilages may occur (Figure 6-8). In type I, the larynx is of normal diameter but the entire trachea, or much of the trachea, is narrowed (1 to 3 mm diameter in the newborn) to a point just above the carina. The main bronchi are often normal in diameter, but may be more transverse than usual and frequently malacic. In some cases, the bronchi are also stenotic and composed of “O” rings of cartilage. In type II, the trachea begins with normal diameter, but then funnels down over a variable length to a tight stenosis. The location of the funneling and of the tight stenosis varies widely. It may be located proximally with a more distal normal segment. In many cases, the stenosis is long, involving more than half of the trachea. In type III,
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6-5 Congenital web at cricoid level. The arrow marks the thin mucosal structure. FIGURE
A
B
C
D
Congenital tracheal stenosis. General categories of stenosis are diagrammed. Types I–III redrawn from Cantrell JR and Guild HC.16 A, Type I: All or most of the trachea is stenosed. B, Type II: Funnel stenosis variously located and of variable length. C, Type III: Short segmental stenosis, sometimes below an anomalous right upper lobe bronchus. D, Type IV: Anomalous right upper lobe bronchus with a “bronchus” to horizontally branching bronchi to the rest of the lung. The right upper lobe bronchus is at the normal carinal level. The bridge bronchus is stenotic, and lesser stenosis may involve part of the trachea above. In some cases, the trachea is elongated as shown, but the upper lobe bronchus is absent. Circles indicate locations of left pulmonary artery sling when present. FIGURE 6-6
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segmental stenosis may occur at any level in the trachea and be of any length, but it occurs most often in the lower trachea. Bronchial anomalies may be present, such as a misplaced right upper lobe bronchus (bronchus suis), which takes off from the trachea above an area of segmental stenosis. Tracheal stenosis may also be present above an anomalous right upper lobe bronchus, with tighter stenosis or “bridge bronchus” below the lobar bronchus. The right main bronchial anomaly is also found as an isolated variant apart from congenital stenosis, and is then usually closer to the carina. With some frequency, patterns are seen which manifest variable groupings of funnel tracheal stenosis, bronchus suis, a “bridging bronchus” (often the most severely stenotic segment), and distal branching into horizontal bronchi to right lower lobe and left lung, forming an inverted “T” (Figure 6-9).17 The point of relatively transverse bronchial branching is held not to be the true carina, because the trachea plus “bridging bronchus” to this point is much longer for age than normal and contains many more rings than the normal trachea. These patients are more difficult to correct surgically than patients with stenosis in a trachea with a more normal pattern. Repair is described in Chapter 33A, “Repair of Congenital Tracheal Lesions: Tracheoplasty for Congenital Tracheal Stenosis.” In over half of the patients, congenital tracheal stenosis may be accompanied by many other malformations, including cardiac anomalies, hyaline membrane disease, pulmonary anomalies, inguinal hernias, imperforate anus, radial aplasia, and megaureters. Segmental stenosis of the distal trachea may be associated with an aberrant left pulmonary artery, the so-called “pulmonary artery sling” (“ring sling complex”) (see Figure 6-9).18,19 The left pulmonary artery originates from the proximal portion of the right artery and passes behind the trachea to the left lung (Figure 6-10). In this course, it indents, but rarely obstructs the
B 6-7 Congenital tracheal stenosis with complete cartilage rings. A, Resected specimen of stenotic segment. The regular ring structure is evident. The ends flare toward normal diameters. B, Photomicrograph of cross section of trachea, demonstrating the complete “O” ring of cartilage. FIGURE
A
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FIGURE 6-8 Congenital stenosis with irregular, confluent plates of cartilage, a very rare malformation. A, Resected specimen. Note the absence of visible rings externally, in comparison with the usual structure of congenital stenosis shown in Figure 6-7A. A cross-sectional specimen below clearly shows completely circular cartilage. B, Luminal surface of A. Note the irregular “haustra” of disordered cartilage plates covered with mucosa.
B A
esophagus. In most of these patients, completely circular “O” rings of cartilage are found in the stenotic segment. The length of tracheal stenosis most often extends beyond the region of the anomalous pulmonary artery sling. Where stenosis is not present, there may instead be a malacic segment at the level of the artery.19 The artery alone can also obstruct the right main bronchus. Division of the anomalous pulmonary artery and reimplantation into the main pulmonary artery anterior to the trachea fails to relieve the airway obstruction when stenosis or malacia are present.19,20 The ligamentum arteriosum, which effectively makes this a ring, is also divided. Sometimes, it has been possible to resect the stenotic tracheal segment and shift the artery anteriorly prior to anastomosing the trachea, as suggested might be possible by Grillo.21,22 This is desirable because of a high rate of stenosis of reimplanted
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6-9 Congenital stenosis with associated left pulmonary artery sling, the ring sling complex. A, A short segment distal stenosis is present. B, A bridge bronchus is adjacent to the anomalous artery. The apparent length of “trachea” is much greater than that in A, and the bronchial branching approximates an inverted “T.” FIGURE
A B
pulmonary arteries in children. The anatomic disposition of the pulmonary arterial sling, however, does not generally permit this transposition without division and reimplantation of the anomalous artery. The distortion of the artery may also compress the resected trachea and cause recurrent obstruction.23 Congenital tracheomalacia that is not the result of compression by vascular structures, by an extrinsic mass, or related to congenital TEF, occurs only rarely. It is infrequently documented in convincing fashion. Primary congenital tracheomalacia is recognized early in life, sometimes in previously apparently healthy infants. It is characterized by progressively noisy respiration, a “seal-bark” cough, episodic cyanosis, increased respiratory rate, intercostal retraction, and stridor which is most noticeable on expiration.24,25 Symptoms worsen with agitation and respiratory infections. Apneic spells may occur. Fluoroscopy may be of diagnostic help but bronchoscopy (preferably rigid) is the key diagnostic technique, showing tracheal narrowing from front to back, indistinct tracheal rings, and expiratory collapse of distal trachea and main bronchi (Figure 6-11 and Figure 3 [Color Plate 12]). Symptoms of primary tracheomalacia often clear by the second year of life, with stability of the trachea then seen on bronchoscopy. In severe cases, prolonged stabilization of the airway is obtained by tracheostomy. Growth of cartilage eventuates in recovery in most cases, in 2 or 3 years. In others, silicone stents (Y stent for lower trachea and main bronchi) can provide long-term patency. Expandable stents are not advisable because of possible permanent tracheal injury and growth problems.
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FIGURE 6-10 Anatomic relationships with anomalous left pulmonary artery sling. A, Anterior view showing the aortic arch and ligamentum arteriosum. Note position of vagus and recurrent laryngeal nerve. B, Transverse-sectional view of the left pulmonary artery sling behind the trachea. A = aorta; BC = brachiocephalic artery; E = esophagus; LCC = left common carotid; LPA = left pulmonary artery; LS = left subclavian artery; PA = pulmonary artery; T = trachea; V= vagus nerve.
A
B
Focal malacia may also occur in relation to a widened membranous wall or residual pouch after repair of TEF and esophageal atresia (Figure 6-12). Defective cartilaginous rings may also be found at this level. Filler and colleagues considered this association to be the most common cause of tracheomalacia in infants, although precise explanation is lacking.26 If severe collapse follows TEF repair, aortopexy may have to be considered.27 Since gastroesophageal reflux can also be present, antireflux surgery may be needed. In a rare case, agenesis or hypoplasia of the right lung may result in mediastinal displacement and rotation severe enough to result in compression of the remaining bronchus at its origin, which is analogous to postpneumonectomy syndrome (see Chapter 15, “Tracheobronchial Malacia and Compression”).28,29 When a short segment of malacic trachea accompanies pulmonary artery sling rather than segmental congenital stenosis, it may be managed by tracheopexy (or aortopexy) in conjunction with division and reimplantation of the anomalous left pulmonary artery.30 Laryngomalacia quite often causes inspiratory, fluttering stridor in the newborn, but usually corrects itself in 1 or 2 years, although it may require tracheostomy—which in turn may lead to later tracheal stenosis. Since this book does not pretend to give comprehensive coverage of laryngologic problems, no more will be mentioned of congenital laryngeal lesions such as absence or deformities of laryngeal cartilages, cysts and laryngoceles, congenital nerve paralysis, or of hemangioma and lymphangioma of the larynx, the latter an extension of cervical cystic hygroma. Tracheobronchomegaly (Mounier-Kuhn disease) appears to be of congenital origin, but most patients have not become overly symptomatic until midlife.31,32 Then, symptoms can often be traced back to youth. It has also been discovered in children. The condition is very rare and thought possibly to be due to absence
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B
6-11 Congenital tracheomalacia causing difficulty early in life. Malacia of the lower 45% of the trachea and of the left main bronchus was identified at 6 months. At age 12, a 12 mm silicone Y stent was placed to allow the patient to be more active. A 14 mm Y stent was fitted at age 15. Minor granulation tissue in the left main bronchus was removed and that limb of a new stent was shortened at age 19. The stent continues to be well tolerated. Resection and primary reconstruction would be too hazardous. The lack of visible cartilage discourages posterior wall splinting, and the location deters the use of external circumferential splinting. A, Bronchoscopic view at carina. Note the absence of a visible ring structure in the anterior tracheal wall. The left main bronchus shows collapse. The right maintains its patency. B, Tomogram demonstrating lower trachea and left main bronchus, held patent by a silicone Y stent. Also, see Figure 3 in the Tracheobronchial Endoscopic Atlas (Color Plate 12). FIGURE
of the trachealis muscle. The anterior tracheal wall may become indented as the rings fold backward (see Chapter 15, “Tracheobronchial Malacia and Compression”). Vascular rings are associated with tracheal and esophageal compression and, if of long enough duration, with tracheomalacia. Tracheal compromise is seen most commonly with double aortic arch, right aortic arch with retroesophageal left subclavian artery and left ligamentum arteriosum, and right arch with mirror image brachiocephalic artery and left ligamentum (Table 6-1) (Figure 6-13). The multiple patterns of vascular rings that occur have been well described and will not be cataloged here again.33,34 Right aberrant subclavian artery from left aortic arch may be associated with dysphagia due to esophageal compression (dysphagia lusoria), but infrequently causes tracheal symptoms. Aneurysm of an aberrant artery late in life, however, can cause tracheal compression. In infants, severe respiratory symptoms may result from vascular rings, with stridor, “crowing,” and repeated respiratory infections. Dysphagia and aspiration may occur. Diagnosis was traditionally made by barium esophagogram. However, vessels are now identified by computed tomography (CT) with contrast or by magnetic resonance imaging (MRI). Division of the ring and ductus or ligamentum arteriosum plus excision of a Kommerell’s diverticulum, if present, usually serves to correct the basic constriction.35–39
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6-12 Symptomatic tracheal diverticulum remaining after repair of a congenital tracheoesophageal fistula. Although the cartilaginous rings were not malacic, the combination of a patulous membranous wall and a diverticular pouch created obstruction. A, Bronchoscopic view of superior (arrow) and inferior lips of the wide-mouthed diverticulum, both of which extend obliquely across the field of view. The distal trachea is above and the patulous membranous tracheal wall below the slit-like mouth of the diverticulum (arrow). B, Diverticulum exposed by sternotomy. Penrose drains encircle and retract the trachea, allowing the posterior diverticulum to protrude (arrows). Forceps point to the diverticulum. A vascular loop encircles the brachiocephalic vein. The diverticulum was excised and the membranous wall was closed in a linear fashion, pulling the splayed rings into normal configuration to provide a normal tracheal lumen. Symptoms were relieved. FIGURE
An aberrant subclavian artery may be best divided and reimplanted or possibly just divided in an infant. Continued tracheal obstruction after surgery may be due to malacia and require intubation or splinting. This must be differentiated from residual obstruction due to an unresected aortic diverticulum or continued vascular compression, as described below.
Table 6-1 Congenital Vascular Rings Which Obstruct Trachea Anomaly
Double aortic arch
Right arch, retroesophageal Left subclavian artery Left ligamentum arteriosum
Right arch, mirror image brachiocephalic artery Left ligamentum arteriosum
Mayo Type33
IA
IIIB
IIIA
Reference 33 34 35 36 37
19 36 17 61 11
4 13 1 34 11
4 3 18 18 4
144
63
47
57
25
18
Total %
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FIGURE 6-13 Principal vascular rings which may compress trachea. A, Mayo type IA: double aortic arch, dominant right arch. B, Mayo type III B1: right aortic arch, left ligamentum arteriosum, retroesophageal left subclavian artery. C, Type IIIA: right aortic arch, left ligamentum arteriosum, mirror image left brachiocephalic artery. Ligamentum may connect to the brachiocephalic artery or to the upper descending aorta. Adapted from Stewart JR et al.33 A = aorta; E= esophagus; LA = ligamentum arteriosum; PA = pulmonary artery; RCC, LCC = right, left common carotid arteries; RS, LS = right, left subclavian arteries; V = vagus nerve.
C
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A variant of vascular right tracheobronchial compression has been observed in a small number of patients with right aortic arch and a right descending aorta, ligamentum arteriosum, and anomalous left subclavian artery with a Kommerell’s diverticulum at its origin (Figure 6-14). The aortic arch may be high, and bends sharply in a “hairpin” configuration as it descends either to the right of or directly over the vertebral spine, leaving little space beneath the arch for the airway (Figure 6-15). All patients with this anomaly had chest wall abnormalities, which markedly narrowed the space between the sternum and vertebrae. Previous division of the ligamentum, excision of the aortic diverticulum, and division of the anomalous subclavian artery in some patients, in different combinations elsewhere, had failed to relieve airway compression. Two patients who had failed to improve after the procedures mentioned above, obtained relief only after division of the aortic arch with pexy of the ends following bypass grafting from the ascending to descending aorta (see Chapter 32, “Surgery for Tracheomalacia, Tracheopathia Osteoplastica, Tracheal Compression, and Staged Reconstruction of the Trachea”). In a third patient, division of ligamentum, excision of the aortic diverticulum, transposition of the aberrant subclavian, and arch aortopexy sufficed. This was an initial corrective operation without prior surgery. If, however, severe malacia has already developed in the compressed airway, additional measures such as tracheal splinting or resection may be needed. A fourth patient with such malacia, after multiple prior operations, needed a tracheobronchial stent. These problems appear to be highly individual. The general principle of correcting cardiovascular lesions as early as feasible may offer the additional advantage of preventing severe tracheomalacia. Backer and colleagues reported 8 children with a similar anomaly, who had recurrent respiratory symptoms and/or dysphagia, despite prior division of a left ligamentum arteriosum in 7 of the children.40 All had a Kommerell’s diverticulum still present. Resection of the diverticulum and reimplantation of a retroesophageal left subclavian artery into the left carotid artery relieved tracheal compression. The aorta is presumed to have descended on the left in these patients and no chest wall deformity was described. Konstantinov has noted, as we did, that symptoms are not always relieved by this limited procedure, although he found this to occur primarily with an encircling right aortic arch which descended on the
6-14 Right aortic arch, right descending aorta, aberrant left subclavian artery, and Kommerell’s diverticulum. The airway compression is not necessarily relieved by excising the aortic diverticulum and transplanting the aberrant subclavian because of the narrow space between ascending and descending aorta. Left ligamentum arteriosum is usually present to the diverticulum or LS. E= esophagus; PA = pulmonary artery; RCC, LCC = right, left common carotid arteries; RS, LS = right, left subclavian arteries.
FIGURE
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FIGURE 6-15 Tracheal obstruction by “hairpin” aortic arch: right aortic arch, right descending aorta, anomalous left subclavian artery originating from aortic diverticulum. A, Aortagram showing a high and narrow arch with a large Kommerell’s diverticulum (arrow) from which the left subclavian artery originates. B, Computed tomography (CT) scan showing the trachea (arrow) compressed and displaced by the right aortic arch. Note the short distance between the sternum and vertebral bodies. C, Postoperative CT after aortic bypass, division of arch, and pexy of ascending arch by a Goretex sling (arrow) passed around an anterior rib. The tracheal lumen is widely opened.
left.41,42 Intraoperative bronchoscopy can monitor the degree of correction achieved by successive procedures, beginning with lesser ones, as in one of our patients cited above. Compression of the trachea by a prominent innominate artery has been relieved by pexy of the artery and aortic arch to the sternum, leaving the trachea attached so that it is pulled forward and suspended.43,44 The validity of this diagnosis has been questioned, but there are instances of obstruction relieved by surgery.45 The artery has been divided and reimplanted more proximally on the aorta to successfully remove the point of tracheal compression and also the possibility of recurrence following suspension.46,47
Acquired Lesions Postintubation stenoses are preponderant among acquired airway lesions in children.48 Unfortunately, these injuries include laryngotracheal stenoses resulting from the preferential use in this age group of endotracheal tubes, often uncuffed, for ventilatory support. Laryngotracheal stenosis is much more difficult to correct than a purely tracheal lesion. In a study of 854 newborns who required intubation and ventilation (1979–1983), Marcovich and colleagues discovered a 0.6% incidence of severe subglottic stenosis, where
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uncuffed, small diameter tubes had been used nasotracheally.49 As in adults, the length of the period of intubation does not seem to be critical, although the period of risk is obviously greater with prolonged intubation. The reported incidence of injury has varied widely, and it remains difficult to define specific conditions that produce sufficient erosion to cause stenosis in one patient and not in another (see Chapter 11, “Postintubation Stenosis”). Tracheostomy, used either by choice to limit the period of endotracheal intubation or as treatment for laryngeal or subglottic obstruction resulting from endotracheal intubation, may produce its own stenotic or other obstructive lesions in infants and children. The uses of carefully placed vertical incisions in cartilage and specifically designed pediatric uncuffed tracheal tubes for tracheostomy in the infant have reduced the incidence of stomal tracheal stenosis (see Chapter 10, “Tracheostomy: Uses, Varieties, Complications”). Obstruction also occurs, although less often, by compression deformity of the soft tracheal wall of infants, just proximal to the stoma, due to pressure by the curvature of the tube. This is correctable by use of T tubes, which, however, may be difficult to care for in infants due to their tiny diameter. Overall, nasotracheal intubation has more often been selected for prolonged ventilation in these age groups. A small number of postintubation tracheoesophageal fistulae have been seen, and, fortunately, only a rare tracheo-innominate artery fistula (see Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula” and Chapter 13, “Tracheal Fistula to Brachiocephalic Artery”). Post-traumatic stenosis from external injury is uncommon except in older children. Stenosis due to inhalation burns is difficult to treat because of the basic pathology of burned tissue (see Chapter 9, “Tracheal and Bronchial Trauma”). Tumors, benign or malignant, occur uncommonly. In 198 primary tracheal tumors, only 4 were in patients under the age of 10 years, and 8 occurred in patients between 11 and 19 years of age.50 These included granular cell tumor, fibrous histiocytoma, neurofibroma, mucoepidermoid tumor, carcinoid (most common), adenoid cystic carcinoma, and rhabdomyosarcoma. Other tracheal tumors noted in childhood were fibrosarcoma, squamous cell carcinoma, hemangiopericytoma, and chondroma. “Congenital” tumors compressing the trachea or bronchi included cystic hygroma, hemangioma, teratoma, and thymic cyst.51 Recurrent respiratory papillomatosis due to the human papilloma virus causes wart-like excrescences in the larynx and trachea, in children as well as in adults. Patients present with hoarseness, a weakened cry, cough, respiratory infections, choking, and obstruction of progressive severity. The disease is vertically transmitted in children. Present treatment is repeated laser vaporization. Multiple antiviral adjuvants have been tried, including interferon. Although most children enter spontaneous remission, recurrences do emerge. Malignant degeneration has rarely been reported in children.52 Hemangiomas of the airway are frequently associated with cutaneous hemangiomas of the chin, lips, mandibular region, and neck.53 In the subglottic region, proliferation produces hoarseness and stridor. Respiratory failure can follow, often at between 6 to 12 weeks of age. Management includes corticosteroids and tracheostomy since the lesions usually regress spontaneously. Cryotherapy and laser therapy have been used, but scarring and stenosis can follow, especially if the subglottic lesion is extensive. Rarely, an extrinsic lesion such as a bronchogenic cyst or mediastinal tumor will produce compression of the trachea or secondary malacia in early childhood.54 For example, apparent malacia over a long tracheal segment, seen in a 21⁄2-year-old child, proved to be due to compression by a large thymic cyst (Figure 6-16). Because of prolonged compression of the trachea by the cyst, which had apparently been present since birth, removal alone did not immediately restore a satisfactory airway. Splinting with a tracheostomy tube was required until the cartilages became firm with further growth. Rarely, a bronchogenic cyst at the carina may produce compressive symptoms in children. Adenocarcinoma was discovered in the base of an obstructing cyst in an 8-year-old girl, and curative carinal resection was secondarily performed (Figure 6-17). Obstruction of the trachea or bronchi has resulted from infection with tuberculosis, histoplasmosis, diphtheria, rhinoscleroma, and syphilis (see Chapter 14, “Infectious, Inflammatory, Infiltrative, Idiopathic,
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6-16 Thymic cyst compressing the trachea in a 21⁄2 -year-old boy. A, The lateral roentgenogram demonstrates tracheal deformation. B, The cyst as it was being extracted from the mediastinum via cervical incision. FIGURE
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and Miscellaneous Tracheal Lesions”). All except histoplasmosis (in adults) are vanishingly uncommon in developed countries. Bulky tuberculous enlargement of mediastinal or hilar lymph nodes can compress and erode the relatively soft trachea or bronchi of children.55 Subcarinal nodes are commonly involved.
FIGURE 6-17
Computed tomography scan in an 8-year-old girl, showing a bronchogenic cyst deforming the carina, but with irregular protrusion into the lumen, which proved to be adenocarcinoma in the base of the cyst. After secondary carinal resection, she remained disease free in 16-year follow-up.
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Sequelae include pulmonary collapse or hyperinflation, airway perforation, bronchostenosis, and caval or esophageal obstruction. Glandular enlargement can occur despite antituberculosis drug treatment, and indeed has been observed to likely increase on initial treatment. Surgical treatment is reserved for symptomatic patients. Acute obstruction may demand emergent surgery. Enucleation and curettage of obstructing nodes and of caseous material, suture and muscle flap repair of a bronchial opening, and conservation of all but destroyed lung are effective.56 Lymph node excision is avoided in order to minimize complications. Late fibrous bronchostenosis is best treated by segmental bronchial resection and anastomosis with preservation of the lung, unless it is irretrievably damaged. Corticosteroids are unlikely to improve gross airway obstruction. Pneumonectomy has become a rare procedure in childhood, so that postpneumonectomy syndrome, once thought to occur principally in childhood, is now seen largely in adults (see Chapter 15, “Tracheobronchial Malacia and Compression”).
Assessment Clinical Findings The diagnosis of congenital tracheal stenosis and other obstructive anomalies is based on a high degree of suspicion in infants and children with respiratory distress. Inspiratory and/or expiratory stridor may be present, accompanied by retraction. Recurrent or persistent cough and exercise intolerance occur. There may be a history of respiratory difficulties of lesser intensity since birth, or shortly after birth, or of repeated and stubborn respiratory infections. Strangely, dyspnea may be episodic. Cyanosis and apneic episodes may occur. In some cases, difficulty in intubation had led to the diagnosis. Late manifestations of congenital stenosis may represent the child’s respiratory demands outpacing the ventilation permitted by the stenotic airway. Only then may a clinical history be retrospectively traced to a much earlier time. Other obstructive lesions are manifest in similar ways. The clinical presentation of congenital TEF is described in Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula.” Caution about delayed manifestation of small congenital H-type tracheoesophageal fistulae and bronchoesophageal fistulae must be repeated. Acquired stenosis due to intubation for ventilation is signaled by shortness of breath on exertion and stridor in the wake of a history of intubation, usually for respiratory support, with or without tracheostomy. Children diagnosed with “asthma,” who fail to respond to treatment, must be suspected of an organic airway lesion. If the child has been previously ventilated, the working diagnosis should be airway stenosis, until proved otherwise.
Imaging Air tracheograms and careful fluoroscopy provide precise information about the presence of an anomaly, its location, and type (see Chapter 4, “Imaging the Larynx and Trachea”) (Figure 6-18). Contrast media are usually unnecessary and may cause complications when used in tiny airways. Tomograms offer specific additional detail, but in many hospitals are no longer available of useful quality. CT scanning provides precise information on the cross-sectional area and extent of lesions, with three-dimensional reconstruction available. An advantage of spiral CT is the rapidity of examination, which is important in small children. MRI offers similar information (Figure 6-19) and is especially useful to delineate associated cardiovascular anomalies. The role of cardiac catheterization to delineate such lesions has thus diminished. MRI may be difficult to accomplish in infants and small children. Angiography is also used less often, but it still provides a “gold standard” for precise and complete delineation of vascular anomalies. Echocardiography is also very helpful to identify cardiovascular anomalies. Left pulmonary artery sling must be identified preoperatively
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6-18 Imaging of congenital tracheal stenosis in an 11-year-old boy. A, The simple oblique roentgenogram shows the location (arrow) and length of stenosis as well as the length of normal trachea from larynx to carina—all critical information for the surgeon. B, Postoperative view of the same patient. Note the increased acuteness of the carinal angle. FIGURE
because its approach and anesthesia can be different from that in a patient who has an isolated congenital tracheal stenosis. Barium swallow is still an important diagnostic method in identifying a vascular ring or aberrant subclavian artery. If a child has a tracheostomy in place, it is preferable to obtain initial roentgenograms with the tube temporarily removed, if the patient can and will tolerate removal. A physician who is able to replace the tube promptly must be in attendance, with adequate preselected equipment on hand. Fistula to the esophagus requires fluoroscopy, with careful administration of a small volume of contrast medium to judge the exact location and approximate size of the communication. If a pulmonary artery sling has been previously corrected by reimplantation, a perfusion scan or pulmonary angiogram should be considered, since this vascular anastomosis in infants has a tendency to stenose and, at best, perfusion will be diminished on the left side. The information could be critical since operative correction of a tracheal stenosis might otherwise be conducted with left main bronchial intubation, thus ventilating an unperfused or underperfused lung.
Endoscopy Careful use of a flexible pediatric bronchoscope can clarify much about a lesion. The bronchoscope should not be passed into a tightly stenotic lesion in order to avoid causing edema and inflammation, which might precipitate acute obstruction. Indeed, in a significantly symptomatic child, in whom the presence of stenosis is already known from radiologic study (even if definition is not wholly complete), bronchoscopy is usu-
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B
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D
6-19 Congenital stenosis in a 31⁄2-year-old boy. A, Sagittal magnetic resonance study showing narrowed supracarinal segment. B, C, and D clarify tracheal diameters in a normal segment, at the beginning of cartilaginous “O” rings and at maximum stenosis. FIGURE
ally best deferred to the time of planned surgical repair. This also applies to less critical patients. Surgeons must always do the bronchoscopy themselves, irrespective of any prior examinations. Definitive bronchoscopy, ideally performed just prior to a planned surgical procedure for correction of the lesion, is best accomplished with rigid Storz ventilating pediatric bronchoscopes. The 3.5 mm rigid bronchoscope (OD 5.7 mm) will not pass through a tiny stenosis. However, either a flexible pediatric bronchoscope (2.7 mm) or a long telescope (OD 2 mm) allows a more distal examination. These may be inserted through a larger rigid (ventilating) bronchoscope seated proximally, or through a pediatric operating
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laryngoscope. Bronchoscopes should not be forced into a stenosis, nor should any attempt be made to dilate the narrowing, if it is congenital. Ventilation is maintained by intermittent placement of an endotracheal tube into the laryngoscope. Fine suction devices are necessary. Circular “O” rings of cartilage, found in many cases of congenital stenosis, are clearly visible bronchoscopically, if looked for (Figure 6-20A and Figure 1 [Color Plate 11]). Disordered or fused cartilages are present in congenital stenosis, but far less often (Figure 6-20B and Figure 2 [Color Plate 11]). As in adults, the larynx must be examined, especially in the presence of a postintubation lesion. The glottis is visualized in passing with the rigid bronchoscope and telescope. However, the pediatric Holinger laryngoscope, used in conjunction with a telescope, provides a superior view of laryngeal structures. If any complexity is identified, consultative examination by a pediatric otolaryngologist is advisable. Cooperation in the initial endoscopic examination of these patients is beneficial for all. Tracheoesophageal fistula is identified in the membranous wall of the trachea (Figure 6-21). Instillation of methylene blue-colored saline into the esophagus via a high placed nasogastric tube may conclusively identify a small H fistula. Confusion can result if a large volume of dyed saline refluxes above the cricopharyngeus and spills over the arytenoids into the airway. Esophagoscopic identification of a fistula may be more difficult unless the fistula is large or distended by a cuffed tube in the trachea.
Treatment and Results Healing of the Juvenile Trachea: Growth and Tension The question of whether anastomosis performed in the juvenile trachea will grow adequately has been answered positively. As an example, Maeda and Grillo experimentally demonstrated that tracheal anastomosis in puppies resulted in some narrowing at the anastomotic site in the adult, caused by infolding of adjacent rings and restriction of growth by cicatrization of the membranous wall (Figure 6-22).57 At the anastomotic site, the average growth was 82% of normal sagitally, and 75% coronally, values equivalent to
A
B
FIGURE 6-20 Bronchoscopic observations in congenital stenosis. A, Complete cartilaginous “O” rings are easily identified visually. B, Very rarely, irregular cartilaginous walls such as in this case are seen in a stenotic segment. Resected at age 9. See also Figures 1 and 2 in the Tracheobronchial Endoscopic Atlas (Color Plate 11).
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6-21 A, Congenital H-type tracheoesophageal fistula, visualized bronchoscopically posteriorly. B, Esophageal opening of the same fistula, on the anterior wall. The dark area below is the esophageal lumen. (Courtesy of Dr. Hon Chi Suen.)
FIGURE
20% stenosis of the tracheal lumen. Tension on the anastomosis caused by resection of progressively longer segments of trachea led to greater anastomotic narrowing when performed in puppies rather than in adult animals.58 Puppies were more susceptible to tension than adult animals. Levels allowing safety from disruption were 1,000 g in puppies and 1,750 g in adult dogs. The range of permissible resection was about half that of the adult, about 25% of the entire length of the juvenile trachea. Within permissible tension limits, fully adequate airways resulted (Figure 6-23). The use of a few spaced sutures from tracheal rings proximal and distal to the anastomosis, to reduce tension on the suture line, lessened postoperative narrowing.59 This is effectively accomplished clinically by our present use of lateral traction sutures (see Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection”). Other laboratory work has generally confirmed this postanastomotic tracheal growth, with variations probably being due to experimental design.60–62 As experience with tracheal resection and reconstruction in infants and children increased, clinical results confirmed these expectations of anastomotic growth.63–66 Couraud and Monnier and their colleagues observed growth following similar laryngotracheal procedures.67,68 This contrasted with a diminished tracheal diameter and the need for later revisions or resection in children treated endoluminally or by plastic procedures. Further evidence of lesser tolerance of anastomotic tension in the juvenile trachea has been adduced, confirming the desirability of confining resection in children to 25 to 30% of the tracheal length.69 Growth of tracheal cartilage appears to occur continuously on the convex or outer side of a ring, with resorption taking place on the concave surface, without identifiable growth centers.70 Personal serial bronchoscopic observations of unoperated congenital stenosis showed that the stenotic segment grew proportionally to the normal trachea. Manson and colleagues demonstrated growth radiologically.71 These observations led to the expectation that growth would occur after slide tracheoplasty. Macchiarini and colleagues observed growth in an experimental model approximating slide tracheoplasty for long congenital stenosis.72 Such growth has now been clinically confirmed by Grillo and colleagues.73
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FIGURE 6-22
Growth of trachea after experimental division and anastomosis. A, Differences in cross-sectional shape in a puppy, a 7-monthold dog that underwent anastomosis as a puppy, and an adult dog. Differences are minor in those of adults. B, Circular narrowing at the anastomotic site after full growth. The lumen remains completely adequate. C, Measurements of internal and external diameters and wall thickness (mm) at and above anastomoses after growth. Reproduced with permission from Maeda M and Grillo HC.57
Non-Operative Treatment Growth has also provided numerous instances where lesser acquired stenotic lesions or limited areas of malacia improved or became sufficiently corrected over time to obviate need for reconstruction. For this reason, and since anastomosis is more safely performed in larger airways, I have approached many pediatric patients conservatively, even accepting prolonged presence of a tracheostomy tube in the interim. Although a silicone T tube has many functional advantages over tracheostomy for prolonged interim treatment, the failure rate of T tubes in very small tracheae is much greater than in the adult, due to easy obstruction of the tube.74 A particularly favorable application of the T tube is in a child with obstruction from a residual depressed flap in the anterior tracheal wall due to the tracheostomy tube lying just below. A T tube splints the deformed wall flap forward, allowing cartilage to remodel in the corrected position. The tube is later removed and the stoma allowed to close. Permanent stents of small caliber or expandable
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FIGURE 6-23
Diagram of tracheal cross sections at anastomotic sites when fully grown, from puppies that were transected or resected, and in dogs resected as adults. Sagittal diameters are vertical (cm). Fully adequate airways resulted within the range of permissible resection. Reproduced with permission from Maeda M and Grillo HC.58
stents should be avoided, because of their failure to account for growth, their ease of occlusion, their potential to cause additional injury, and the difficulty of removal. Dilation of acquired stenosis, just as in the adult, provides temporary relief at best in most patients, with or without use of the laser. An exception may be web-like congenital strictures (rarely seen as acquired lesions). Kim and Hendren noted satisfactory results in selected cases with electrocautery excision of subglottic webs.75 Congenital segmental stenosis should not be dilated since this can split the “O” rings, which are usually present.
Resection and Reconstruction of the Trachea Increasingly, tracheal resection and anastomosis, both for acquired lesions and short congenital stenoses, has been performed with considerable success (Figure 6-24A). Procedures developed for adults have served well in children, but principally for acquired stenosis, since the length of most congenital stenoses precludes resection and reconstruction. Warnings about intolerance of greater anastomotic tension and danger of postoperative obstruction due to edema and secretions remain valid. Technique, as ever, must be precise and meticulous. For anastomosis in children, I prefer using interrupted 5-0 Vicryl sutures because of their ease of handling, strength, and lack of granulation formation. Conclusions from experiments purporting to demonstrate the superiority of polydioxanone monofilament sutures (PDS) over Vicryl braided sutures, were based on a scale of early histologic reaction to sutures and not on long-term results.76 In large numbers of cases, Vicryl sutures have been shown to be as close to an ideal tracheal anastomotic suture as has yet been offered, in terms of ease of use, strength, minimal reactivity and, most importantly, absence of long-term complications such as granulomas, suture erosion into the lumen, and anastomotic separation and stenosis.77 PDS is somewhat more difficult to handle and has no positive advantages to highlight its recommendation. Anesthetic management by the cross-field intubation technique, similar to that used in adults, has worked extremely well in skilled hands. Some cardiac surgeons, however, used to using cardiopulmonary bypass in infants, feel that this simplifies their operative field. My principle has been to avoid possible complications from unnecessary additional procedures.
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Carcassonne and colleagues published their experience in pediatric tracheal reconstruction in 1973, and Nakayama and colleagues reported reconstruction in infants and small children in 1982, updating the series in 1990 to 45 patients.63,6678 Grillo and Zannini described the management of obstructive tracheal disease in children in 1984, including 20 who underwent resection and reconstruction: 3 for congenital stenosis, 9 for postintubation stenosis, 1 for TEF, 2 for post-traumatic stenosis, 1 for idiopathic stenosis, and 4 for primary tumors.64 Two had carinal resections. One death occurred in the congenital group and one in the acquired group. Seventeen enjoyed good results. Gaissert and colleagues extended these observations to carinal and main bronchial reconstruction.79 The series was updated in 2002.69 Success of over 80% in 73 major operations was considerably less than in adults, however.
Repair of Subglottic Laryngotracheal Stenosis When the subglottic larynx and, usually, the adjacent upper trachea is stenotic, most often due to ventilation through an endotracheal tube, correction is more difficult than when the problem is purely tracheal. A simple sleeve resection is not possible if the recurrent laryngeal nerves are to be saved, just as in the adult. Conservative measures, such as repetitive dilation, local and systemic steroids, laser or electrocoagulation ablation, and prolonged stenting, have only occasionally been successful. A host of complex operative procedures usually associated with laryngofissure, buccal, cutaneous, or cartilage grafts with stents, anterior and posterior cricoid splits, and castellated incisions, have also produced variable results. Despite failure rates of 20 to 50% in some series of such procedures, satisfactory success with the cricoid split and cartilage graft has been reported.48,80,81 However, more recently, the effectiveness and safety of primary resection and anastomosis have been acknowledged.82 Single stage methods of repair (see Chapter 25, “Laryngotracheal Reconstruction”) involve resection of the anterior cricoid arch and stenotic scar anterior to the posterior cricoid plate, with preservation of the posterior plate and perichondrium to protect the recurrent nerves, tailoring the distal trachea to reconstruct the subglottic airway and to resurface the posterior cricoid plate. Developed initially for adults, these techniques have been applied successfully in children, with considerable success.83–89 Monnier and colleagues described 15 children so treated, with 14 successes.90 Three were of congenital origin and 12 resulted from intubation. Decannulation was achieved in a single procedure in 14 of the patients. Follow-up in 10 of the children from 5 to 14 years showed good laryngeal growth despite removal of the anterior cricoid.68 Couraud and colleagues noted a similar experience, also with laryngeal growth.67 More recent experience further supports this approach, which seems on a rational basis to be superior to the cricoid split with cartilage graft insertion, leaving the stenosis in place.
Congenital Stenosis In 1982, Kimura and colleagues described anterior patch tracheoplasty for treatment of congenital stenosis involving the entire trachea (see Chapter 33, “Repair of Congenital Tracheal Lesions”).91 The technique followed the principle of Gebauer’s wire reinforced dermal grafts for tracheal and bronchial stenosis.92 Stenosis too long to be treated by resection and anastomosis was incised vertically throughout its length and the tracheal diameter widened by fitting a long cartilaginous graft (Figure 6-24B). An endotracheal tube was left in place until the patch became firm. A later report augmented their experience.93 Idriss and colleagues modified the procedure by using pericardium, which required not only postoperative splinting with an endotracheal tube but suture suspension of the pliable pericardial patch to mediastinal structures as well.94 Considerable difficulty was encountered, with granulation tissue formation, necessitating multiple postoperative bronchoscopies (mean 3.8), especially for grafts extending far distally (mean 16).95 Twenty-one patients underwent pericardial patch repair, with 2 operative deaths and 3 late deaths. Six needed later tracheostomy, 2 for airway stenting. Troubled by the lack of intrinsic sup-
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FIGURE 6-24 Surgical techniques for treatment of congenital tracheal stenosis. A, Resection and reconstruction, suitable only for shorter stenosis. B, Patch tracheoplasty. Incision of long stenosis and widening of trachea with free cartilage or pericardial grafts.
port, a later modification proposed by this group was anterior division of the stenosis, and excision of a central piece of trachea to be used for part of the “gusset,” augmented with pericardium. This complicated repair offers little over simple slide tracheoplasty, described below. Heimansohn and colleagues also used pericardium in a group of 12 patients, and noted only 2 patients with granulations and requiring re-operation of the 10 patients who survived long term.96 Importantly, normal growth of the trachea was noted in longer-term results (1 to 11 years, mean 5 years).97 In 2 patients examined postmortem 13 and 18 months, respectively, after pericardial patch tracheoplasty, it was found that the pseudostratified columnar epithelium had lined the dense mature collagenous scar tissue that had replaced the pericardial patch.98 Jaquiss and colleagues returned to cartilage patch tracheoplasty, with granulation and dehiscence in 1 of 6 patients, all of whom survived.99 Mechanical ventilatory support was provided for a mean of 11 days, with a median postoperative hospitalization of 17 days. In time, cartilage grafts are also replaced by mature scar tissue and reepithelized by ciliated columnar epithelium.100 The luminal enlargement is maintained. Linear sections of cadaver trachea, chemically fixed so that the tissue is not viable, have been used as a patch to widen long stenoses of various etiologies.101 Both the biological processes of healing and the benefits of this technique compared with the use of native tissues are unclear (see Chapter 45, “Tracheal Replacement”).
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Slide tracheoplasty (see Chapter 33, “Repair of Congenital Tracheal Lesions”), first proposed by Tsang and colleagues and successfully employed by Grillo, meets the problems of long segment congenital stenosis by using tracheal tissue alone to widen the lumen, giving stability, minimizing granulation tissue formation, and assuring more likely prompt healing.102,103 A complete epithelial surface is also immediately provided. The stenosis is divided horizontally in its midpoint, and the upper and lower segments of stenosis are incised vertically through their entire extent, one anteriorly and one posteriorly. Corners are trimmed and the two segments slid together for suturing (Figure 6-24C). The circumference of the trachea is doubled and the cross-sectional area approximately quadrupled (Figure 6-25). Since the ends of the cartilaginous walls tend to curl inward, a lobulated cross-sectional appearance of a figure 8 character may be produced, so that the area is slightly less than quadrupled (Figure 6-26). Since the affected segment of the trachea is halved in length, even stenosis of the entire length of trachea does not preclude the procedure. This operation was performed in 8 patients ranging in age from 9 days to 19 years (Figure 6-27).73 Five were done with simple cross-field ventilation and 3 with a period of bypass necessary to reimplant a divided pulmonary artery sling, to perform other cardiac procedures or because of myocardial disease. Segments resected were between 36 to 83% of tracheal length. Two had anomalous right upper lobe bronchi. Only 1 required 3 days of intubation for ventilatory support, principally for airway clearance. Hospitalization ranged from 8 to 13 days. One minute granuloma appeared late and was removed bronchoscopically. Follow-up between 1 and 10 years shows good growth of the reconstructed trachea in the younger patients.73 Functional results, which can be measured in older patients, are excellent (Table 6-2) (Figure 6-28).
FIGURE 6-24 (CONTINUED) C, Slide tracheoplasty, reconstructing the trachea with (unresected) native trachea, providing fourfold widening of the lumen. Probably preferable even for most shorter stenoses as well as for long stenosis.
C
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A
B
FIGURE 6-25 Bronchoscopic views before (A) and after (B) slide tracheoplasty in a 3-month-old boy with stenosis of one-half of his tracheal length. In A, “O” rings are clearly visible. In B, the widening of the lumen allows the carina to be seen distantly. The irregular margins of the tracheoplasty are evident. The child was completely relieved of severe symptoms and his trachea grew well.
B 6-26 A, Roentgenogram of a long congenital stenosis treated by slide tracheoplasty. B, Preoperative and post slide tracheoplasty computed tomography scans of the patient. The reconstructed trachea has a bilobed appearance due to the natural curvature of the tracheal wall segments. The enlargement is therefore less than fully fourfold, but it fully relieved obstructive symptoms. FIGURE
A
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201
6-27 Application of slide tracheoplasty. Diagrams of our first 8 patients, with percentages of trachea affected by stenosis treated by this technique. In number 6, the lesser supracarinal stenosis did not require correction, and the slide procedure was applied to the “bridge” bronchus. True tracheal length naturally varied with age. Reproduced with permission from Grillo HC et al.73
FIGURE
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Table 6-2 Pulmonary Function Before and After Slide Tracheoplasty for Congenital Stenosis Patient 1 Variable FEV1 (L) VC (L) FEV1/VC PEFR MBC RV/TLC
Preoperative (%)
Patient 2
Postoperative (%)
Preoperative (%)
Postoperative (%)
1.26 (40)
1.51 (52)
3.0 (79)
3.37 (93)
1.3 (37)
1.68 (47)
4.10 (92)
4.28 (102)
0.97 (107)
0.90 (110)
0.74 (85)
0.79 (91)
2.3 (37)
3.18 (51)
4.39 (63)
5.4 (80)
50 (40)
44 (40)
121 (79)
135 (93)
0.62 (238)
0.52 (217)
0.28 (93)
0.13 (44)
FEV1 = forced expiratory volume in 1 second; MBC = maximum breathing capacity; PEFR = peak expiratory flow rate; RV = residual volume; TLC = total lung capacity; VC = vital capacity.
FIGURE 6-28 Flow volume loop in a 19-year-old patient who underwent slide tracheoplasty, preoperatively and 1 year postoperatively. She was totally relieved of symptoms.
The results of slide tracheoplasty suggest its superiority over patch tracheoplasty.73,103 Since reports from three major children’s hospitals show an average incidence of only one or two such cases yearly in each institution, a long time may be needed to establish a preferable method statistically. Nevertheless, a number of recent reports have indicated a growing acceptance of slide tracheoplasty.73 I recommend this technique even over resection and reconstruction, if there is any likelihood of too much tension after resecting a lesion of borderline length. It should be unnecessary to point out that this technique has no application in acquired fibrous stenosis, where the absence of a “normal” tracheal wall would result in restenosis.
Congenital and Acquired Tracheal Lesions in Children
Resection and reconstruction of congenital stenosis is still the method of choice for short segments of stenosis, where lack of anastomotic tension may be confidently predicted. Resection is also uniquely applicable in the case of a short bridging bronchus, especially where maximum narrowing is located at one end of the stenotic bridging bronchus. Cantrell and Guild described this procedure and I have found it useful.16,73 Extreme caution must be used in designing the exact size and location of the anastomotic apertures proximally and distally, to avoid angulation of the bronchi (see Chapter 33, “Repair of Congenital Tracheal Lesions”). Transplantation of trachea remains impractical at present. Problems of maintenance of viability of the epithelium and cartilage, tissue rejection, and hazards of prolonged immunosuppression are discussed at length in Chapter 45, “Tracheal Replacement.” A further problem with any form of transplant in juveniles, as well as with synthetic or composite grafts to replace lost trachea, is the need for growth as the child grows. Experimenters tend to forget this in their enthusiasm for “new” technology. For similar reasons, I believe that permanent or semipermanent expandable stents are to be avoided in the juvenile trachea, if the disease is not soon likely to be lethal. In addition, such stents can irreversibly damage the trachea and prove to be nearly irremovable. Silicone inlying stents, such as the Dumon stent, should be used with measured judgement. I have surgically corrected children who were seemingly condemned to indefinite periodic replacement of Dumon stents. Silicone stents cause reactive granulomas and thus extend the original pathology. The inadvisable path of repeated laser treatments should also be avoided.
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CHAPTER SEVEN
Primary Tracheal Neoplasms Hermes C. Grillo, MD
Characteristics Clinical Presentation Diagnostic Studies Treatment and Results
Primary tracheal neoplasms are still often diagnosed long after the onset of symptoms or signs, particularly in the absence of hemoptysis. Benign neoplasms may be unrecognized for many months or even for several years. The duration of symptoms for malignant lesions prior to diagnosis is 6 to 18 months, reflecting more rapid growth, and especially, onset of hemoptysis. Even pulmonologists remain unfamiliar with tracheal tumors because of their rarity and a corresponding dearth of information about their occurrence and behavior. Reassured by a chest radiograph, which is often read as normal, the physician assigns the reason for wheezing and exertional dyspnea caused by a tumor to “adult onset asthma” or to chronic lung disease. Inappropriate treatment, once the lesion has been recognized, is an equally serious problem. It is still not widely appreciated that modern techniques of tracheal surgery, combined with radiotherapy, can produce cure or long-term palliation in many patients with primary malignant tumors.1–6 Overall results of treatment are more encouraging than oncologic results of management of carcinoma of the lung, to which major therapeutic efforts are regularly directed. As with most tumors, the best opportunity for cure presents at the time of diagnosis. The physician who deals with a primary tracheal tumor should, therefore, be fully aware of its potential clinical course and the best methods available for treatment. If expertise in management is not available in the physician’s area, the patient should be referred to an appropriate center. Patients continue to be seen with resectable tracheal tumors that are presumed on the one hand to be of limited malignancy or on the other hand to be incurable and that are treated by transbronchial ablation or irradiation alone. Laser treatment has been employed repetitively on resectable tumors—a treatment that can never cure a malignant tracheal neoplasm and only rarely a benign one. When such patients are seen many months and sometimes years after initial diagnosis, longitudinal spread, particularly of adenoid cystic carcinoma, has become so extensive that surgical resection and reconstruction is too often no longer possible. High-dose “curative” irradiation administered without initially considering surgical excision may make later resection prohibitively risky. Even benign tumors present increasingly greater technical problems for definitive surgical resection if their management has been delayed by ill-conceived attempts at local management—often with a laser. Simple microscopic exam-
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ination makes it clear that only full-thickness ablation of the wall of the trachea, which contains the base even of most benign tumors, could possibly lead to extirpation (see Figure 3-12 [Color Plate 2] from Chapter 3, “Pathology of Tracheal Tumors”). Unfortunately, at present in the United States, considerations of cost and jurisdiction under managed care can keep patients from proper treatment. This could worsen if the web of managed care and capitation spreads.
Characteristics The incidence of primary tracheal tumors in the general population is not precisely known. Ranke and colleagues found 2 patients with tracheogenic carcinoma in 1,744 cancer deaths.7 Culp noted 4 patients with primary tracheal tumors in 89,600 autopsies.8 It is not a surprise that the diagnosis is seldom considered, even by pulmonologists. The majority of primary tracheal tumors in adults are malignant. Thirty-six percent of a series of 198 patients with primary tracheal tumors seen at the Massachusetts General Hospital (MGH) over a 26-year period presented with primary squamous cell carcinoma (SCC) and 40% with adenoid cystic carcinoma (ACC).1 The remaining 24% of the total included 9 other malignant lesions, 17 of intermediate character, such as carcinoid and mucoepidermoid tumors, and 21 were clearly benign. None of the SCCs were secondary to laryngeal, bronchogenic, or esophageal carcinoma. By 2002, the number of ACCs and of SCCs seen had risen to 135 each, a total of 270 patients in these categories alone. The very wide variety of tumors other than SCC and ACC is noteworthy (Table 7-1). The pathology of primary tracheal tumors is reviewed in Chapter 3, “Pathology of Tracheal Tumors,” and illustrated in a color fascicle. Bronchoscopic views of some tumors are also to be found in the color section. Tracheal tumors are even rarer in children. Twothirds are benign, since of the most common adult tumors, ACC is only occasional in children and SCC is nearly unique. In a review of the literature and from their own experience, Desai and colleagues found only 38 children with tracheal tumors over a 30-year period.9 Over half were diagnosed initially as “asthma”; 39% were more than 50% obstructed when diagnosed. Hemangiomas and granular cell tumors were most common in the benign category, and mucoepidermoid tumors and histiocytomas in the malignant. Malignant tumors usually appeared in adolescence. Primary SCC of the trachea may be exophytic or ulcerative, localized or longitudinally infiltrating, or less commonly, may show multiple areas of involvement scattered throughout the trachea (Figures 7-1, 7-2 and Figures 4 and 5, Color Plate 12). Invasive squamous cancer may also be found deep within what appears to be an area of papillomatous change, which on superficial biopsy reveals apparently in situ carcinoma. If such a lesion is grossly visible, it often does have deeper areas of invasive carcinoma. As SCC grows, it extends longitudinally and circumferentially in the tracheal wall, and may penetrate extraluminally to involve adjacent structures. Tumors may occur at any level of the trachea or carina. Adjacent recurrent laryngeal nerves and the esophagus may be invaded directly. The most common sites of metastases are adjacent peritracheal lymph nodes. Hematogenous metastases to the lung, bone, liver, or adrenals are less common initially. Age and gender incidences of SCC of the trachea are similar to those of carcinoma of the lung (Table 7-2), with peak incidence between 50 and 70 years, predominating in males. The etiologies seem to be identical. Except in 4 cases, every patient we have seen with SCC of the trachea has been a cigarette smoker, usually for many years. One exception had received arsenicals for dermatologic treatment in youth and suffered multiple squamous skin cancers on exposure to sunlight. Another had worked for many years with a multitude of organic chemicals and had previously suffered squamous carcinoma of his tongue base. In 2 other nonsmokers, no etiology was determined, although the extent of involvement of the overlying thyroid gland in 1 raised a question of whether that tumor might have been a primary squamous carcinoma of the thyroid invading the trachea. Forty percent of our patients with SCC
Primary Tracheal Neoplasms
Table 7-1 Primary Tracheal Tumors other than Adenoid Cystic and Squamous Carcinomas Benign Squamous papilloma Multiple Solitary Pleomorphic adenoma Granular cell tumor (myoblastoma) Glomus tumor Fibroma Fibrous histiocytoma (pseudotumor, plasma cell granuloma, xanthoma) Lipoma Leiomyoma Hamartoma Chondroma Chondroblastoma Schwannoma Neurofibroma Paraganglioma Hemangioma Hemangioendothelioma Vascular malformation
Malignant Adenocarcinoma Adenosquamous carcinoma Small cell carcinoma Basaloid squamous cell carcinoma Atypical carcinoid Malignant fibrous histiocytoma Melanoma Chondrosarcoma Spindle cell sarcoma Rhabdomyosarcoma Fibrosarcoma Leiomyosarcoma Kaposi’s sarcoma Lymphoma Lymphoepithelial carcinoma Angiosarcoma
Intermediate Malignancy Carcinoid Mucoepidermoid Plexiform neurofibroma Pseudosarcoma Plasmacytoma Acinic cell carcinoma
of the trachea who underwent resection had either a previous history, a concurrent finding, or a later occurrence of SCC of the respiratory tract.1 Cancer arose in the tongue, tonsil, larynx, trachea (second primary lesion), and lung. Basaloid squamous cell carcinoma, a rare and aggressive variant consisting of basaloid cells with either dysplastic epithelium or in situ squamous epithelium or invasive tumor, usually occurring in the upper aerodigestive tract, may be found in the trachea and may be deeply ulcerative, invasive, and metastatic.10 It may be confused with ACC histologically and with neuroendocrine carcinoma. Adenoid cystic carcinoma may appear deceptively benign. Its former appellation, “cylindroma,” masked its truly malignant character.1,11 Indeed, it used to be described clinically and pathologically under a general heading of “bronchial adenoma.” This included a heterogeneous collection of “cylindroma,” carcinoid, and mucoepidermoid tumors plus a few true adenomas. Both terms are best abandoned. Adenoid cystic carcinoma of the trachea occurs over a wide age range, from the twenties through the seventies (see Table 7-2). Distribution between male and female is quite even but with female predominance (72 to 63 in 135 patients). No relationship has been discerned with cigarette smoking or other known carcinogenic factors. Thirty-three percent were smokers compared with at least 66% of patients with SCC. Although the
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B
A FIGURE 7-1
Squamous cell carcinoma of trachea. Bronchoscopic views. A, Small tumor in the upper trachea. B, Large tumor at the carina.
B 7-2 Varied gross presentations of squamous cell carcinoma (SCC) of trachea. A, Tomographic delineation of a small exophytic lesion in a 60-year-old man. The “dome” of the subglottic larynx lies superiorly. The tumor (arrow) lies a few rings below the cricoid cartilage. B, Surgical specimen of A. An overlying lobe of thyroid was also removed in-continuity to provide a better lateral margin. Four years later, the patient underwent a right lower lobectomy for SCC of the lung. He died 20 years later without recurrence of either carcinoma. FIGURE
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7-2 (CONTINUED) C, Smooth-appearing exophytic lower tracheal squamous carcinoma in a 56-year-old male. Right upper lobectomy was done 15 years later for squamous carcinoma. Neither tumor recurred. D, Tomogram showing a large squamous lesion of the lower trachea (between arrows) in a 55-year-old male . The lower open arrow marks the carina. E, Surgical specimen from D. The base of tumor was less extensive than gross tumor and provided a microscopically clear margin. F, Ulcerating squamous cell carcinoma in a 57-year-old man. Adjacent lymph nodes were involved. Three years later, he developed pulmonary metastases and a second primary squamous carcinoma of the tongue. FIGURE
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Table 7-2 Incidence of Primary Tracheal Tumors by Age Number of Tumors Age (years) 1–10 11–19 20–29 30–39 40–49 50–59 60–69 70–79
Squamous — — 1 1 9 29 24 6
Adenoid Cystic
Other
— — 13 16 19 15 13 5
4 8 11 9 5 6 4 1
Adapted from Grillo HC and Mathisen DJ.1
tracheal tumor is histologically indistinguishable from that found in salivary glands, it is indeed rare for a single patient to present with tumors, either synchronous or metachronous, both in the salivary gland and in the trachea. The airway tumor may occur at any level of the trachea, but seems to be more prevalent in the lower trachea and carina. It occurs much less often in main bronchi and very rarely distal to that. Multiple tumors are extremely rare. Adenoid cystic carcinoma presents grossly in a variety of ways (Figures 7-3, 7-4, 7-5, and Figures 7 through 11 [Color Plates 12, 13]). The lesions may be red and rubbery in appearance or granular and friable. One or all borders may appear sharply defined or the tumor may infiltrate diffusely. Frequently, adjacent to the main mass of tumor, which clearly projects from the mucosa, there is evidence of tumor infiltration beneath the mucosa or in the tracheal wall. The margins are often indistinct when examined with a magnifying telescope through a rigid bronchoscope. Mucosal elevation or friability is seen and new tumor vessels may be identified in the mucosa. Commonly, these cancers invade microscopically submucosally and perineurally for long distances beyond grossly visible disease. Microscopic pathology is described in Chapter 3, “Pathology of Tracheal Tumors.” The lesion may be essentially circumferential within the trachea or may involve both main bronchi at the carinal level. Proximal tumors may involve the larynx in varying degrees. The extratracheal tumor mass may become bulky. Although these tumors may grow to considerable size extramurally into the mediastinum prior to recognition, they often displace adjacent structures before invading them, including the esophagus and pulmonary artery. I have seen the left pulmonary artery shown to be completely occluded by the pulmonary angiogram, but at operation found only compression, without invasion of the pulmonary arterial wall. The esophagus is more likely to be directly invaded by a posterior tumor, especially in its muscular wall. A recurrent laryngeal nerve may be defunctioned by direct invasion. Adjacent paratracheal regional lymph nodes on both sides may be involved, but apparently not as frequently as by SCC of the trachea. For a tracheal tumor, these should be considered as N1 nodes. Late local recurrence of ACC, even 15, 20, or more years after apparent cure by surgery and irradiation, is a discouraging characteristic. Despite almost uniformly excellent early response to external irradiation treatment, recurrence usually follows if irradiation alone is used for treatment. Recurrence is seen at the prior site of the maximum bulk of tumor, usually between 3 to 7 years following treatment. Improved long-term outcome seems to follow combined surgical resection and full-dose radiotherapy, but these results must be regarded with reservation in view of the tumor’s indolent clinical behavior and the small amount of information about long-term behavior.6,11,12 Metastases to the lung occur all too frequently, but
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7-3 Adenoid cystic carcinoma. Bronchoscopic views. A, Tumor invading the subglottic larynx in a 54-year-old man, at and above the cricoid level, with essentially circumferential extension. Resection with laryngeal salvage was impossible. The patient elected palliative neutron beam radiation to preserve laryngeal speech. Tumor recurred, as expected, in 6 years. B, Adenoid cystic carcinoma confined to midtrachea, 2 × 2.7 cm, in a 64-year-old man. The tumor, 4.5 cm from the carina, was removed by cervicomediastinal tracheal resection. C, Tumor at the carina and in the left main bronchus, also extending into the proximal right main bronchus. Left carinal pneumonectomy was done via a “clamshell” incision. FIGURE
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may emerge late and enlarge slowly. Patients may remain asymptomatic for many years with multiple enlarging pulmonary metastases. Indeed, such behavior may justify surgical removal of a tracheal lesion despite the presence of numerous lung metastases, in order to prevent airway obstruction. Experience is small, but excision of pulmonary metastases has almost never produced cure or seemed to alter the course of the disease. Usually, metastases are too numerous for complete removal. Where a few metastases have been resected, other metastases have usually appeared later. Metastases to bone and other organs also occur. In a minority of cases, although no unusual histologic characteristics were identified, ACC proved to be rapidly and widely aggressive. Manifestations in these few patients included local invasion of the central pulmonary artery, intrapericardial extension, multiple pleural metastases, and multiple rapidly-growing pulmonary or osseous metastases. The primary tumor in such a patient is not necessarily large.
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FIGURE 7-4 Radiologic presentation of adenoid cystic carcinoma (ACC). A, Small, well-defined tumor of the upper trachea, delineated by tomography. The vocal cords and subglottic larynx are well outlined. The involved right wall of trachea is deformed. The right recurrent laryngeal nerve was involved. This 25-year-old man underwent cervicomediastinal resection and also received 5,000 cGy of irradiation postoperatively. He remained well and without recurrence 30 years later. B, Tomographic detail. Broad-based ACC in a 40-year-old woman extending into the cricoid cartilage on the left (arrow). The left side of the lower larynx was included in the resection. The left recurrent laryngeal nerve had to be resected. The trachea was bevelled to fit the line of laryngeal resection. Laryngeal structures are visible above. Vocal cords are widely abducted. C, Computed tomography scan in a 39-year-old woman shows the midtrachea encircled by ACC. She was treated by segmental tracheal resection. D, Large tumor at the carina obstructing the left main bronchus totally and the right bronchus subtotally. Left carinal pneumonectomy was necessary.
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7-5 Gross surgical specimens of adenoid cystic carcinoma. A, Tumor with large extratracheal component and involved peritracheal lymph nodes. B, A 6.5 cm resection for an extensive tumor of the midtrachea in a 43-year-old man. Complete sternotomy plus extension into the right thorax was necessary for removal. Laryngeal release was also required. A tentative initial distal incision in the trachea was made, but was too close to the tumor. C, Open specimen of B, showing the intraluminal extent of tumor. D, Another extensive tumor in a 27-year-old man, involving distal trachea, carina, and left main bronchus. Left carinal pneumonectomy was necessary via bilateral thoracotomy, with elevation of the right main bronchus to proximal trachea. Laryngeal release helped because of the high level of anastomosis. Irradiation followed. The patient is now 22 years after resection. FIGURE
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Over 40 years, between 1962 and 2002, 135 patients with ACC and 135 with SCC were seen at MGH.6 In the second 20-year period, 97 with ACC and 98 with SCC were seen. Of these, 101 (75%) with ACC and 90 (67%) with SCC were resected. A miscellaneous group of other primary tracheal carcinomas has also been encountered, usually with so few examples of each type that little can be said about general behavior patterns. These neoplasms include adenocarcinoma in the carina of a child at the base of a bronchogenic cyst. Carinal excision following prior removal of the cyst at another hospital led to cure. Adenosquamous carcinoma was encountered, involving both the trachea and the lower larynx, and was treated by cervical mediastinal excision with mediastinal tracheostomy. Another such tumor, involving the right main bronchus and carina, was treated successfully (15-year survival) by carinal resection and right upper lobectomy with anastomosis of the bronchus intermedius to the left main bronchus and the left main bronchus to the trachea. Two instances of small cell carcinoma of the trachea were encountered, reconfirmed on pathological review, with no involvement of adjacent lung and no nodal metastases. These patients remained disease free over many years after surgical resection plus conventional adjuvant therapy for small cell carcinoma. The rarity of primary tracheal small cell carcinoma was observed elsewhere.13 A greater frequency of small cell carcinoma in the trachea, which was described in the past, probably included secondary invasion from the lung.14 Lymphoepithelial carcinoma (Schmincke tumor), more often occurring in the nasopharynx, has been seen in the trachea.15 Treatment was resection and irradiation. It appears that melanoma can occur primarily in the trachea. As in the patient reported by Duarte and colleagues,16 our single patient had undergone removal of a small cutaneous lesion, without histologic record, many years before. Lower tracheal resection was done. However, the patient had no subsequent recurrence of melanoma in 13-year follow-up. Except for 11 patients with carcinoid tumor of the trachea and carina, which we noted in 1978, carcinoid has not often been reported in the trachea.17 Behavior of carcinoid in the trachea and carina appears to be much the same as in the bronchi, where it is much more common (Figure 7-6, and Figures 12 through 15 [Color Plate 13]).18 The rarer atypical variety may be highly malignant. Typical carcinoids, when resected with limited but negative histologic margins, did not recur. In these cases, no regional nodes were involved. Carcinoid syndrome is seen only rarely with bronchial carcinoid, often, but not uniformly, in the presence of hepatic or other metastases. One patient with carcinoid syndrome from a tracheal tumor was referred with massive mediastinal lymph node metastases following resection of an atypical tracheal carcinoid. Block removal of lymph nodes temporarily controlled the syndrome until tumor recurred. Carcinoid appears to respond poorly to irradiation. We have not encountered Cushing’s syndrome with tracheal carcinoids, possibly a reflection of the low incidence of this hormonal effect in these tumors.19 Histological examination of the wall of the trachea from which a typical carcinoid tumor arises usually demonstrates deep enough origin that it is impossible to remove the tumor definitively intraluminally with a laser or by other means (see Figure 3-12 [Color Plate 2] from Chapter 3, “Pathology of Tracheal Tumors”; Figure 7-6F). Only transient relief of obstructive symptoms can be obtained by debulking the tumor within the lumen. Full-thickness excision of the tracheal wall is required for complete removal. On the membranous wall, a carcinoid tumor of moderate size often extends through the posterior wall. Claims of cure appear to be either an illusion or such a rarity that harm may be done by encouraging delay in accomplishing simple curative surgical extirpation. Mucoepidermoid tumor presents in the trachea and main bronchi, with malignant behavior in a small proportion of cases in most series.20,21 In Heitmiller and colleagues’ series from MGH, 3 of 18 patients with mucoepidermoid tumors of the trachea and bronchi were high grade and fatal within 16 months.21 All the others survived (Figure 7-7). Nodal spread was infrequent in low-grade tumors. Two tracheal tumors were treated by sleeve resection, one by laryngotracheoplastic resection, and another underwent carinal
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C FIGURE 7-6 Carcinoid tumors. A, Bronchoscopic view. Carcinoids are often smooth, rounded, and vascular in appearance. B, Irregular carcinoid located just above the carina. C, Radiographic details of the carina of a 31-year-old woman treated for “asthma” with progressive dyspnea for over a year. Anteroposterior view on left radiograph and lateral view on right radiograph. Both show the lower tracheal tumor clearly.
pneumonectomy. Bronchial sleeve resection sufficed for another 5 patients, and lobectomy was needed in 7. Jensik, Faber, and colleagues made similar observations earlier. Irradiation treatment of high-grade tumors seemed ineffective.20 In 31 children and adolescents, in whom the tumor was located in the bronchi, prognosis appeared to be better than in adults, with no recurrence seen after resection alone.22
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7-6 (CONTINUED) D, Computed tomography scan of the patient described in C. E, Gross specimen of the patient in C, exhibiting a typical carcinoid in character and appearance. F, Carcinoid invading the carina in a 19-year-old female. Operative photograph shows protrusion of tumor through the membranous wall of the trachea. The lefthand tape encircles the trachea. The two on the right encircle the right main (above) and left main (below) bronchi. The futility of laser treatment, which she had on several occasions, is obvious. FIGURE
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Two pleomorphic adenomas in the trachea were excised in our series (Figure 7-8, and Figure 18 [Color Plate 13]). These tumors are rare, may occur in children, and produce obstructive symptoms. They are mixed tumors of salivary gland type with varied mixtures of epithelial and stromal components. By 1992, 19 cases had been gathered.23 Similar tumors in the bronchus were first described by Payne and colleagues.24 Complete surgical excision and reanastomosis is the proper treatment. One of our patients had a similar tumor excised years before from a salivary gland, but there was no evidence then or since of metastatic disease. It was, therefore, classified as a primary tracheal tumor. Malignant features evidently occur, but were not present in these patients.
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FIGURE 7-7 Mucoepidermoid tumors. A, Lateral neck roentgenogram in a 28-year-old woman showing a posterior lesion (arrows) involving the subglottic larynx and upper trachea. Laryngoplasty was done, advancing the posterior membranous tracheal wall to resurface the posterior larynx after tumor excision. Both recurrent nerves remained intact. B, Carinal resection for mucoepidermoid tumor in a 34-yearold man, viewed from the tracheal end of the specimen. Twelve year follow-up was without recurrence.
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FIGURE 7-8 A, Bronchoscopic view of pleomorphic adenoma. This true adenoma of the trachea is very rare. B, Another pleomorphic adenoma, with areas of adenocarcinoma present.
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Squamous papilloma of the trachea may occur as a solitary, moderate-sized lesion, which should be removed by segmental tracheal resection. Naka and colleagues in 1993 found reports of 5 solitary tracheal papillomas, 7 in the main bronchus, and 41 in a lobar bronchus.25 They proposed surgical resection for wide-based or poorly-defined tumors or for suspected malignancy. Tracheal polyps of inflammatory origin which occur as solitary lesions may also be obstructive.26 Multiple squamous papillomatosis also occurs in many locations in the trachea and at the carina. A fortunately-rare form produces multiple papillomas throughout the distal tracheobronchial tree, with obstructive septic consequences. In children, squamous papillomas of the trachea behave in benign fashion and tend to regress with adolescence (see Chapter 6, “Congenital and Acquired Tracheal Lesions in Children”).27 Their occurrence is related to human papilloma virus and antiviral treatment has been employed. These tumors were treated by repeated endoscopic removal using, in various eras, electrocoagulation, cryosurgery, and now, laser—all seemingly effective. Multiple lesions in adults, on the other hand, may be verrucous lesions, which cover large areas. The process may be so extensive that surgical resection is not possible. This is one of the few clear-cut indications for repetitive laser treatment. The role of photodynamic therapy remains to be clarified. Invasive carcinoma is not uncommon deep within these lesions. Cartilaginous tumors, including osteochondroma, chondroma, chondroblastoma, and chondrosarcoma, are very rare in the trachea and only somewhat more frequent in the larynx.28 Huizenga and Balogh found 10 cartilaginous tumors in 5,000 laryngeal neoplasms.29 Peak incidence is from 40 to 70 years of age. In the larynx, about 70% arise from the cricoid, most frequently from the posterior plate. The tumor is submucosal and produces obstructive symptoms (Figure 7-9, and Figures 16, 17 [Color Plate 13]). A mass may be palpated in the neck. Biopsy is difficult because the tumor is firm and normal mucosa is likely to be obtained. The recurrent nerve is not initially affected, but voice change occurs due to tumor bulk. Hemoptysis is very rare. These lesions are usually chondrosarcomas of low grade and often are indolent in behavior. Treatment is surgical excision with every effort made to save the larynx. Techniques must be individualized. The reconstructive approach is outlined in Chapter 25, “Laryngotracheal Reconstruction.” With conservative approach to maintain laryngeal function, recurrence must be watched for over a long term, with likelihood of later re-resection necessary years later. Salvage laryngectomy is eventually needed in some cases. With recurrences, atypical areas and dedifferentiation are seen.28,30,31 Chondrosarcoma does not respond to radiotherapy. Tracheal chondral tumors seem to be even less common than laryngeal tumors. Limited experience suggests that these are most common in older males.32 They may vary from benign chondroma to chondrosarcoma of increasing malignancy (Figure 7-10). Lesions may be largely intraluminal or extend through the tracheal wall.33–35 Nonproductive cough is followed by dyspnea, first on exertion and then at rest, with wheezing—especially on recumbency—late in the course. With chest x-rays initially “clear,” the diagnosis of asthma is frequently carried for a long period. A medical student with a tracheal chondroblastoma was retrospectively identified as having had tumor present radiologically for 7 years (Figure 7-11). Cartilaginous tumors are radiologically defined on all imaging modalities. Calcification is frequently seen as well as destruction of normal cartilages, especially in the larynx. A bulky extraluminal mass may be visible. Treatment is surgical excision with tracheal reconstruction. The approach is directed by the location and extent of tumor (see Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection,” Chapter 25, “Laryngotracheal Reconstruction,” and Chapter 28, “Reconstruction of the Lower Trachea [Transthoracic] and Procedures for Extended Resection”). Complete excision is necessary to prevent recurrence and hence should be a patient’s first procedure. Bronchoscopic debulking is not definitive treatment. Further, tumors apparently progress in degree of malignancy or transform from being benign to malignant.36 True cartilaginous tumors are distinguished from hamartomas by the absence of other tissue elements that are seen in hamartomas: lipomatous, epithelial, and lymphoid. Our patients with chondroma
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FIGURE 7-9 Chondrosarcoma of larynx. A, Cervical computed tomography (CT) scan showing the low-grade tumor invading the laryngeal wall, notably the left cricoid cartilage, in a 64-year-old man. Calcification is notable in the tumor. Treated by laryngoplastic resection and reconstruction. B, CT reconstruction showing the extent of tumor. C, Bronchoscopic view of the same tumor. The tumor is covered by mucosa. Note tracheal rings distally on the right.
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and chondroblastoma of the trachea had no recurrence after complete resection with primary anastomosis. A low-grade, bulky chondrosarcoma treated by resection developed multiple, slow-growing pulmonary metastases over many years, but had no tracheal recurrence. Hemangioma of the airway occurs, especially in children, in the subglottic region of the airway.37 Half of these children also have hemangiomas elsewhere. The lesions are generally characterized by proliferative endothelial cells. Although lesions regress with age, they may require treatment because of their location either in the lumen of the airway or as an extrinsic compressive mass. Lymphangioma can on rare occasions similarly obstruct the trachea by pressure. Biopsy is to be avoided. A conservative approach is advised. Treatment has included temporizing tracheostomy to await regression, laser, corticosteroids, and in the past, irradiation. Laser therapy generally leads to remission.38,39 Interferon has shown effect but neurological complications may occur.40
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FIGURE 7-10 Chondral tracheal tumors. Benign chondroma causing nearly total obstruction, in a 69-year-old musician who suffered dyspnea and stridor for 9 months before diagnosis of obstruction by flow volume loop. Tomograms show in A, anteroposterior and B, lateral views, how completely the lesion filled the tracheal lumen. C, The cut surgical specimen shows tumor originating from the anterior tracheal wall and abutting the membranous wall. D, Roentgenogram shows a slowly progressive low-grade chondrosarcoma, which originated in the tracheal wall, markedly displacing the trachea, brachiocephalic vein, and filling the upper mediastinum.
An extremely rare lesion, but quite different, is arteriovenous vascular malformation in the anterior and middle mediastinum, which may present within the tracheal lumen as an obstructive lesion (Figure 7-12). These are not mediastinal hemangiomas, which are most often quite discrete.40,41 The arteriovenous vascular malformation is fed by multiple arteries and entwines the mediastinum with a network of vasculature.40,42 Such malformations are presumed to be congenital and not true tumors. Unless thrombosis has occurred, the vascular endothelium is quiescent, rather than proliferative as in hemangiomas.
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E
FIGURE 7-10 (CONTINUED) E, Gross specimen, excised via a cervicomediastinal approach. Concurrent carotid endarterectomy was performed. F, The same specimen opened. The patient developed pulmonary metastases and died many years later, but without airway obstruction.
F
In 2 patients, the malformation extended from the thoracic inlet to the diaphragm and was fed by multiple arterial branches. These included thyrocervical, internal thoracic, right coronary, a branch of right subclavian, and bronchial arteries. The huge resulting shunt enlarged the azygos vein and the superior and inferior vena cava. The malformation protruded through the wall of the trachea in both, visible bronchoscopically as a plexus of pulsating vessels. One patient had undergone right upper and middle lobectomies in childhood for hemangiomatous lesions not further defined in prior records, and the other patient had had cutaneous vascular lesions removed in childhood. Both were 25 years of age when seen. In one patient, who had severe obstructive symptoms, management was commenced with an extensive program of embolic obliteration of multiple feeding vessels from the neck, mediastinum, and right coronary artery. After
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FIGURE 7-11
Chondroblastoma in a 22-yearold medical student, treated for years for “asthma.” Retrospectively, a tracheal tumor was identified on x-rays taken 7 years earlier. The impression of the endotracheal tube on the soft intraluminal tumor is seen.
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7-12 Arteriovenous vascular malformations (AVM) of mediastinum involving trachea. Angiograms show widespread involvement of the mediastinum and neck. A, A 25-year-old man with protrusion of AVM into the lower trachea (arrow) producing respiratory obstruction. Managed by multiple, repeated embolization of feeding arteries followed by successful tracheal segmental resection. His airway remained clear 19 years later. See text. B, Lateral view of the patient in A. The arrow indicates tracheal invasion. C, A 25-year-old woman with more diffuse tracheal compression by very extensive AVM. Intratracheal protrusion of vessels was also present. See text. FIGURE
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reducing flow in this way, meticulous dissection accomplished resection of the involved portion of the trachea. The basic malformation was not resectable in its entirety. Seven years later, multiple coronary arterial venous fistulae were ligated at another hospital. Since then, the patient has continued to do well. The second patient was not initially sufficiently symptomatic to justify such an extensive and somewhat perilous approach. Years later, she presented at another hospital where embolization failed and surgery was unsuccessful. She died there from intratracheal hemorrhage adjacent to a tracheostomy. Glomus tumor, found most commonly in skin and subcutaneous tissue and especially in fingers under the nails, occurs in the trachea only rarely. It arises from cells related to the glomus bodies, which have smooth muscle characteristics.43 Symptoms in trachea are cough, dyspnea, and hemoptysis. The tumors are benign and treated by limited but complete resection in most cases. Fourteen tracheal cases had been collected by the year 2000.44 The tumors may be very vascular. Initial examination may suggest carcinoid or hemangiopericytoma. Immunohistology clarifies the differential diagnosis. We have also encountered a very diffuse form, which contiguously involved most of the trachea with extension into right and left main bronchi (Figure 7-13). The patient showed early response to external irradiation and brachytherapy, but required a stent to maintain a satisfactory lumen. Long-term palliation cannot yet be assessed, although he continues to do well 31⁄2 years after treatment. Granular cell tumor, formerly termed granular cell myoblastoma, is predominantly believed to arise from Schwann cells, and is of muscle origin (Figure 7-14). The tumors arise most commonly in the tongue and head and neck area. A small number occur in the airways, and most of these are in the larynx or bronchi. Burton and colleagues collected 30 tracheal instances in 1992.45 Age of occurrence was from 6 to 56 years and the tumor was more often found in women. Many patients are black.46 Granular cell tumors can be multiple in the airway or elsewhere, and malignant manifestation is only very rarely described. Tracheal lesions appear most often intraluminally, but can be located or extend extraluminally. Tumor cells are present deep in the tracheal wall in all the larger tumors.47 Endoscopic resection of the smaller tumors has produced some long-lasting clearances. However, the frequency of recurrence (over half), lack of knowledge of the tumor depth in unresected patients, the indolent course of tumor, late recurrences, and the need
7-13 Diffuse glomus tumor. Bronchoscopic view. Widespread nodular disease is evident. Treated by irradiation because of extent. See text. Also, see Figure 19 (Color Plate 14).
FIGURE
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FIGURE 7-14 Granular cell tumor of the upper trachea, posteriorly based and extending over the cricoid cartilage. An arc of posterior laryngeal mucosa and submucosa is visible at the top of the photograph. Only a slit of airway remains anterior to the tumor. The patient, a 14-year-old girl, was critically obstructed.
for very long-term bronchoscopic follow-up after endoscopic removal, favor primary limited but complete sleeve resection of the trachea or bronchus. Patients often present with “asthma,” that is, shortness of breath and wheezing, rather than hemoptysis. One 14-year-old girl with critical obstruction urgently required laryngotracheal resection and reconstruction (see Figure 7-14). Follow-up bronchoscopy revealed a second granular cell tumor in the bronchus intermedius, which was resected without loss of lung tissue. Multiple granular cell tumors elsewhere have also been reported. The question of irradiation hardly arises with this particular pattern of behavior, and when irradiation has been given, its effect has been uncertain. A lesion that occurs infrequently in the lung and sometimes in the gastrointestinal tract is variously termed postinflammatory tumor, pseudotumor, plasma cell granuloma, xanthoma, fibrous histiocytoma, and by composite names formed from these terms (xanthomatous pseudotumor, plasma cell tumor, fibroxanthoma, xanthogranuloma). Histologically, the lesions show proliferative mature plasma cells, reticuloendothelial cells, granulation tissue stroma with proliferative fibroblasts, and contain lymphocytes and fat-laden mononuclear cells. What is still not clear is whether these lesions are true tumors or in fact inflammatory in nature (Figure 7-15). Tumors which might be considered inflammatory on bronchoscopic biopsy may prove to be neoplastic and invasive on final examination.48 Depth of invasion into and through the cartilage varies. Matsubara and colleagues have subdivided these lesions in the lung into three groups.49 They question the relationship of these lesions, which they consider to be of inflammatory origin, with malignant fibrous histiocytoma. In the airway, the tumor causes cough, dyspnea, wheeze, hemoptysis, and pulmonary changes due to obstruction.50,51 Tumors occur at any age but more often in children and young adults. Intraluminal tumor may be polypoid or sessile. Criteria for benignity or malignancy do not seem to be well established.52 The lesions can extend into the mediastinum and the thyroid gland, giving rise to diagnosis of malignant fibrous histiocytoma.53,54 This term is also used to describe high-grade sarcoma, adding to confusion. Laryngeal tumors may more often appear malignant. On the other hand, complete excision of tracheal histiocytoma usually results in
Primary Tracheal Neoplasms
FIGURE 7-15 Inflammatory pseudotumor of the trachea in a 39-year-old woman, extending into the posterior subglottic larynx. A, Endoscopic view. The cricoid is visible anteriorly, tracheal rings distally, and proximal extension of lesion posteriorly. B, Magnetic resonance scan and reconstructions showing localization of the lesion. From left to right: 1) subglottic and linear extent, anterior view; 2) lateral view showing the posterior mass high in the airway; 3) cross section of airway partly obstructed by bulky mass, with thyroid lobes on either side. Removal of tumor required resection of 4 cm of trachea with posterior laryngeal mucosa and submucosa, and sacrifice of invaded right recurrent laryngeal nerve. Temporary tracheostomy was done. There has been no recurrence.
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cure.47,48,53–55 Incomplete excision is likely to result in local recurrence. Increasing the etiologic mystery are reports of resolution of pulmonary lesions following treatment with cortisone or spontaneously.56,57 This last raises a question about the suggested beneficial effect of irradiation or of cortisone.58 In 2 patients with very large pseudotumors of the trachea, excision produced relief without recurrence. Extension of the lesion proximally over the posterior cricoid plate submucosally required excision of the lesion anterior to the cartilage and reconstruction with a tongue of distal tracheal membranous wall (see Figure 7-15). Sleeve resection of an obstructed left main bronchus for another lesion allowed salvage and gradual effective recovery of the chronically inflamed lung. A measured decision must always be made on whether a long obstructed lung can be usefully salvaged. Primary neurogenic tracheal tumors are also uncommon. Horowitz and colleagues found reports of 3 neurofibromas (not with von Recklinghausen’s disease) and 12 neurilemomas (Schwannoma), to which they added another.59 Most were in the lower trachea and produced cough, symptoms of tracheal obstruction, or hemoptysis. Segmental resection was curative. Endoscopic resection may well result in long-delayed recur-
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rence.59 Such a recurrent tumor was dumbbell in form and necessitated carinal resection and reconstruction. Stack and Steckler added an upper tracheal case in 1990, the nineteenth reported patient.60 All were benign. A patient with recurrent plexiform neurofibroma of the lower trachea, following previous local enucleation elsewhere, attained cure (24-year follow-up) by extended resection of the trachea, including a portion of esophageal wall and an isolated area of metastatic seeding in the prior thoracotomy wound (Figure 7-16). Local enucleation of tumors from the tracheal wall is generally not an appropriate method of management. Two patients with tracheal paraganglioma, which involved the posterior wall of the trachea intimately adjacent to the cricoid cartilage, were treated by segmental excision of the upper trachea, removal of a portion of the posterior cricoid plate, and in one, a portion of the muscular wall of the esophagus. There was no local recurrence. One, however, affected with familial incidence of multiple tumors, later presented with bilateral carotid body tumors. His trachea remained clear. This tumor is not common and was first reported in the trachea in 1956.61 Other mesenchymal tumors occur in the trachea. These include leiomyoma, lipoma, and fibroma (Figure 7-17).62–64 Eleven tracheal leiomyomas have been reported, with calcification in one, just as occasionally occurs in esophageal leiomyomas.63 The majority of hamartomatas are found in pulmonary parenchyma, with less than a quarter presenting endobronchially and rarely in the trachea.65,66 Since these lesions rarely produce hemoptysis, the patients often present with a high degree of obstruction after prolonged treatment for “asthma.” In a rare case where a benign connective tissue tumor is based on a narrow pedicle, endoscopic removal alone may produce cure. Such patients, however, should be observed bronchoscopically for years for recurrence. Where there is any degree of involvement of tracheal wall, segmental tracheal resection is advisable. Resection is definitive, and given the limited resection required, carries little morbidity in experienced hands.
A
B
7-16 Plexiform neurofibroma of the lower trachea, in an 11-year-old boy. Resected by enucleation 6 1⁄2 years previously. There was also an incisional recurrence. The patient is tumor-free 24 years after sleeve resection. A, Resected 4.5 cm specimen with surrounding tissue. Lobule of tumor protrudes from the cut end of the trachea. B, Opened specimen showing the extent of recurrent tumor.
FIGURE
Primary Tracheal Neoplasms
FIGURE 7-17
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A, Lateral tomogram of fibroma high in the trachea (arrow). A tracheostomy had been done unnecessarily prior to referral. B, Anteroposterior tomogram showing a low-grade spindle cell sarcoma in the uppermost trachea, just below the cricoid, in a 15-year-old female. The right side of the cricoid was bevelled to provide a wider margin. No recurrence occurred in 10-year follow-up.
A
B
Extramedullary solitary plasmacytomas occur most often in the head and neck. In 1995, Logan and colleagues reported the eleventh such tracheal patient.67 It is not known how likely these patients are to develop multiple myeloma. Resection and subsequent irradiation are advised for the apparently solitary tracheal lesion. Non-Hodgkin’s lymphoma (NHL) is rarely confined primarily to the trachea or bronchi. In addition to lymphocytic lymphoma, mucosa associated lymphoid tissue, and anaplastic histologic subtypes of NHL, lymphoplasmacytoid lymphoma has been found in the trachea.68 Symptoms of tracheal lymphoma are cough and those of airway obstruction, frequently misdiagnosed as asthma. When other anatomic sites are not involved, the disease is often of lower grade histology. Fidias and colleagues identified 5 patients with primary tracheal disease, variously treated with chemotherapy, resection, and irradiation.69 An additional 36 were found to have secondary tracheal involvement. The tumors may arise from mucosal lymphoid tissue, forming “lymphoepithelial” lesions. Since the cases are few, treatment regimens have varied widely. Long-term freedom from disease (12 to 64 months) resulted in 4 of 5 patients followed. One patient required tracheal resection for malacia following complete response to chemotherapy. Irradiation was then added. Maeda and colleagues reported survival over 5 years after tracheal resection only for primary tracheal malignant lymphoma.70 Although experience is sparse, appropriate treatment for stage I extranodal tracheal lymphoma would seem to be chemotherapy, surgery where feasible, and irradiation. Resection may not prove to be necessary in all cases, but when needed, it is best done prior to irradiation. Recurrence may follow after a long interval. Thus, one of my patients, who had tracheal resection with laryngotracheoplas-
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ty for diffuse mixed cell and large cell malignant lymphoma, followed by irradiation, suffered recurrence retroperitoneally 11 years later, without airway disease. Hodgkin’s lymphoma in mediastinal lymph nodes has affected the trachea in late stages from recurrence with perforation (tracheoesophageal or tracheomediastinal fistula) or from fibrous stenosis years after chemotherapeutic and irradiation treatments.71,72 In the latter case, surgical resection seems contraindicated (see Chapter 42, “The Omentum in Airway Surgery and Tracheal Reconstruction after Irradiation”). Fortunately, sarcomas are uncommon. This group includes leiomyosarcoma, fibrosarcoma, and rhabdomyosarcoma.73–75 All are uncommon in the trachea and bronchi. In 1999, Vinod and colleagues discovered in the literature 8 cases of leiomyosarcoma, 6 of malignant fibrous histiocytoma, and 1 each of rhabdomyosarcoma, fibrosarcoma, liposarcoma, and non-human immunodeficiency virus (HIV) Kaposi’s sarcoma, all primary in the trachea.54 Of these 18 cases followed for varied time periods, only 3 died of disease and 1 of other causes. The behavior of sarcomas is variable (see Figures 7-17B, 7-18). Although in our series a low-grade spindle cell sarcoma and a fibrosarcoma did not recur after resection, other sarcomas have been of high malignancy, and, in several cases, have been too extensive for resection when first identified. Irradiation and chemotherapy should probably be considered after surgical extirpation, but data are few for each cell type. In the case of a highly malignant sarcoma, postoperative irradiation has not appeared to delay recurrence. Pulmonary metastases seemed to occur early. Obstructing Kaposi’s sarcoma was also identified exclusively in the subglottic region and trachea of an HIV-positive patient, and treated by laser and irradiation.76 Endobronchial lesions are often present in acquired immunodeficiency syndrome (AIDS) patients with hilar adenopathy and perihilar infiltration, who show Kaposi’s sarcoma of the skin and mucous membranes.77,78 Obstruction may occur. Prognosis is very poor.
Clinical Presentation Patients with tracheal tumors commonly have a long history of persistent coughing, which may steadily worsen. A patient notices gradual onset of shortness of breath on exertion, which will progress to dyspnea
7-18 Infiltrating spindle cell sarcoma with myxoid stroma, involving the lower trachea (T), carina, and much of the left main bronchus (Lt), in a 28year-old man. Carinal resection and left pneumonectomy were followed by 6,000 cGy irradiation and chemotherapy. The hook in the gross specimen is at the carina. The patient died 3 years later of tumor progression. FIGURE
Primary Tracheal Neoplasms
at rest. At this point, the airway is reduced to 30 to 50% of its normal cross-sectional area. Dyspnea may be aggravated by eating and by position. Wheeze is followed by true stridor (Table 7-3).79 Chest roentgenograms are frequently interpreted as normal on the basis of clear lung fields, although the lesion may well be visible if the tracheal air column is examined critically. Unfortunately, a radiologist’s index of suspicion of a tumor in the presence of such symptoms is all too often no greater than that of a pulmonologist or thoracic surgeon. In a patient with signs of upper airway obstruction—dyspnea on exertion, wheeze, or stridor, with or without cough—organic obstruction of the upper airway should be suspected, even if the lung fields appear to be clear on standard roentgenograms. The patients are all too often treated for “adult onset asthma” or for other imprecise diagnoses for long periods of time. Some present with Cushingoid appearance due to prolonged administration of high-dose steroids for treatment of presumed asthma. Such patients should be bronchoscoped early. A benefit of increasing use of computed tomography (CT) scans is earlier identification of tracheobronchial lesions. Hemoptysis occurs sooner or later with many tumors, especially epithelial. It is more common with SCC of the trachea, somewhat less common with ACC and carcinoid tumors, and may not occur at all with many tumors, especially of mesenchymal origin, benign or malignant. Since the warning sign of hemoptysis is less frequent in ACC than in SCC (Table 7-4), the duration of symptoms is usually longer in the former and wheeze has more often developed.6 As expected, resectability diminishes in patients with a longer average duration of symptoms prior to diagnosis in both types of carcinomas (Table 7-5). Hemoptysis mandates bronchoscopy, even with apparently clear lung fields radiologically, and is explained away distressingly often as due to strenuous cough, tracheobronchitis, or pneumonia. The bronchoscopist must think of the possibility of tracheal tumor. In particular, if the tumor is in the upper trachea, the bronchoscopist, even with a flexible bronchoscope, may pass too quickly from the vocal cords to a supracarinal, carinal, and bronchial examination and overlook a small proximal tracheal lesion. If examination is made with the flexible bronchoscope through an endotracheal tube, the tube must be withdrawn to the level of the glottis as the examination is completed so that the entire trachea is visualized. Unilateral or bilateral pneumonitis may occur. Episodes of pneumonia or pneumonitis may respond to treatment, only to recur. Recurrent pneumonia or persisting pulmonary infiltrates, particularly in an otherwise healthy patient, are indications for bronchoscopy. A small tumor low in the trachea may produce recurrent unilateral pneumonitis and yet be of such small size that it is not visualized on tomography. A thin-section CT scan, however, will demonstrate even tiny tracheobronchial lesions. Hoarseness results
Table 7-3 Symptoms and Signs Associated with Tracheal Tumors Number from 84 Patients Dyspnea Hemoptysis Cough Wheezing Dysphagia Change in voice and/or hoarseness Stridor Pneumonia Emphysema and/or asthma
44 28 22 16 13 13* 12 10 9
Adapted from Weber AL and Grillo HC.79 *Eight of 13 had vocal cord paralysis.
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Table 7-4 Comparative Symptoms in Adenoid Cystic Carcinoma (ACC) and Squamous Cell Carcinoma (SCC) of Trachea Number from 135 Patients of each Diagnosis
Dyspnea Cough Hemoptysis Wheeze Stridor Hoarseness Dysphagia Fever Other
ACC
SCC
65 55 29 44 21 10 7 7 12
50 52 60 27 27 13 7 4 14
χ2 0.014 NS < 0.001 0.003 NS NS NS NS NS
Adapted from Gaissert HA et al.6 NS = not significant.
from involvement of a recurrent laryngeal nerve by tumor. Dysphagia is a late symptom caused by esophageal invasion. Untreated, death results from asphyxia, pneumonia, or hemorrhage.
Diagnostic Studies Only very rarely will the imaging studies described in Chapter 4, “Imaging the Larynx and Trachea,” fail to define the location and extent of a tracheal tumor (Figure 7-19). Oblique tomography or thin-section CT scan will usually identify small and otherwise subtle tracheal lesions. Virtual bronchoscopy may add a pictorial dimension, although it is not essential. If pulmonary lesions are also discovered, a fine needle percutaneous biopsy is useful to determine their nature. Bronchoscopy is essential sooner or later (see Chapter 5, “Diagnostic Endoscopy”). The number of tracheal lesions overlooked or diagnosed late will be minimized if bronchoscopy is routinely used in the following circ*mstances: 1) patients who suffer from prolonged cough, dyspnea on exertion, and wheezing or stridor—without precise and proven diagnosis; 2) patients with hemoptysis; 3) patients with recurrent atelectasis, pneumonitis, pneumonia, or persistent unexplained pulmonary infiltrate. Where a lesion has been identified radiologically, is not unusually extensive, and clearly would be best treated by resection, a rigid bronchoscopy is often deferred to the general anesthesia under which resection is planned. The endoscopic management of severe acute obstruction by tumor is outlined in Chapter 19, “Urgent Treatment of Tracheal Obstruction.” If there is possible hazard from an attempt at biopsy and the patient is to be referred to a center for definitive surgical resection, biopsy is best deferred.
Table 7-5 Duration of Symptoms and Resectability Tumor Type
ACC (months)
SCC (months)
Resectable Unresectable
18.3(14.6–21.9) 23.7(15.1–32.3)
4.5(3.6–5.5) 7.6(2.8–12.4)
Adapted from Gaissert HA et al.6 The range is given in parenthesis. ACC = adenoid cystic carcinoma; SCC = squamous cell carcinoma.
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B
A
C
FIGURE 7-19 Radiologic definition of adenoid cystic carcinoma in a 28-year-old woman. A, Standard posteroanterior view shows little obviously abnormal. The arrow indicates tumor in the lower trachea. B, Large lower tracheal mass is seen near the carina on a lateral roentgenogram. C, Anteroposterior tomogram outlines the supracarinal mass.
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E
D
F
FIGURE 7-19 (CONTINUED) D, Lateral tomogram demonstrates the posterior location of the base of tumor. E, Barium esophagogram shows smooth indentation of the esophagus by the mass. F, Computed tomography scan clarifies the component of tumor extending beyond the tracheal lumen. Carinal resection was required for an adequate margin, and was followed by radiotherapy. She remained well and without recurrence 19 years later.
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Functional studies are rarely of much practical use. Obstruction of the central airway requires relief. No matter how diminished the pulmonary reserve is, the patient will be improved by relief of the proximal airway obstruction. The surgical approach to distal tracheal lesions may be altered in the light of very poor pulmonary parenchymal function, however.
Treatment and Results Surgical Management Until the development of current techniques of tracheal resection and reconstruction, prospects for cure of tracheal tumors were small. Even when a lesion seemed potentially curable, some surgeons were often so inhibited by fear of inability to reconstruct the trachea that they settled for very limited local resection, often lateral resection, in an effort to maintain tracheal continuity. Local recurrence frequently followed such limited resection. Radiotherapy almost uniformly resulted in local recurrence even of radiosensitive tumors after varying periods of time, longer with ACC (3 to 7 years) than with squamous carcinoma (1 to 21⁄2 years). Characteristic reports from this era were those by Houston and colleagues in 1969, listing 53 primary tracheal cancers seen over a 30-year period, and by Hajdu and colleagues in 1970, noting 41 patients over a 33-year period.80,81 Because data is limited—due to the still relatively small number of cases reported, the absence of many single institutional series of significant size with prolonged follow-up, and the wide range in types and behavior of tracheal tumors—it is difficult to be categorical about management. However, enough information has accumulated in the past 30 years to formulate preliminary conclusions. In 1978, Grillo reported 63 patients, and in 1990, Grillo and Mathisen reported 198 patients with primary tracheal tumors treated at MGH, the latter report spanning 26 years.1,82 Eschapasse, Pearson, and Perelman, and their colleagues offered significant series.3,4,83 The combined experience of 26 French, German, and Italian hospitals was reported in 1996 by Regnard and colleagues.5 The distribution of primary tumors has been noted previously. In a 1990 series, 132 (66%) of our patients underwent resection with primary reconstruction of the airway (Table 7-6).1 In 9 of these (adenoid cystic 3, squamous 1, other 5), involvement of the lower larynx by a high tracheal tumor required removal of a portion of the larynx with suitable tailoring of the distal trachea to accomplish reanastomosis with a remaining functional larynx (see Chapter 25, “Laryngotracheal Reconstruction”). In an additional small
Table 7-6 Primary Tracheal Tumors (1990) Variable Number of lesions Percentage of total Surgical treatment Excised Percentage of type Explored Resection With reconstruction Trachea Carina Laryngotracheal resection Staged procedure Adapted from Grillo HC and Mathisen DJ.1
Squamous
Adenoid Cystic
Other
Total
70 36 50 44 63 6
80 40 65 60 75 5
48 24 43 43 90 0
198 100 158 147 74 11
41 32 9 1 2
50 22 28 4 6
41 28 13 2 0
132 82 50 7 8
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number of patients (7), the larynx was so extensively involved by a high tracheal tumor that salvage was impossible and laryngotracheal resection was performed (Figure 7-20). Eight staged resections were performed where primary reconstruction was not possible and where it was planned ultimately to reconnect the larynx to the distal trachea in secondary procedures. Since so few of these succeeded optimally, staged procedures were abandoned. Sixty-three percent of all SCCs were resected, as well as 75% of all ACCs and 90% of others. The high resectability rate of the last group was due to the large number of benign lesions (see Table 7-6). Resectability rates remained stable at 68% for SCC and 73% for ACC as the series grew.6 Principal contraindications to resection are 1) extensive linear involvement of the airway such that primary end-to-end resection would not be possible without excessive tension, 2) mediastinal invasion of nonresectable organs, or 3) remote metastases. Although palliative resection may be advisable in some patients with ACC or differentiated thyroid carcinoma, to remove a potentially obstructing lesion even in the face of pulmonary metastases, I usually prefer to embark upon tracheal resection and reconstruction where there is a chance of cure. This applies even more to carinal resection because of increased risks of major surgical morbidity and higher mortality (see Chapter 21, “Complications of Tracheal Reconstruction”). Emergency management of patients with severe obstruction is described in Chapter 19, “Urgent Treatment of Tracheal Obstruction.” If resection and reconstruction is not deemed to be possible because of linear extent of the lesion, primary radiotherapy is employed (see Chapter 41, “Radiation Therapy in the Management of Tracheal Cancer”). Brachytherapy has, on occasion, been added to external beam irradiation in selected patients. In the presence of bulky tumor with extrinsic compression of the tracheal wall, often after failure of radiotherapy, a T tube or a solid or coated expandable stent may span the area of obstruction for a time (see Chapter 40, “Tracheal and Bronchial Stenting”). Surgical approaches (see Tables 7-6, 7-7) and techniques for resection are described in Part 2, “Therapeutic Techniques and Management” (see Chapters 23–25, 28, 29, 34). The differing distribution of tumors is highlighted by the fact that 9 of 41 (22%) patients undergoing primary resection and reconstruction for SCC underwent resection of the carina, whereas 28 of 50 (56%) patients who had ACC were treated by carinal resection. By the year 2002, these figures were 22% and 41%, respectively. Laryngeal release was used 7 times in 82 (8.5%) patients undergoing tracheal resection for tumor. It was earlier also used 5 times in those undergoing carinal resection. I have since concluded that it can be useful in carinal resection, only where a large portion of the trachea itself has been removed, since laryngeal release only assists in advancement of the upper half of the trachea. This was confirmed by Valesky and colleagues in anatomic studies.84 Hilar release, on the other hand, particularly the inferior portion of release at the level of the inferior pulmonary vein, was used in 12 of 32 (38%) patients undergoing transthoracic tracheal resection and 23 of 50 (46%) patients treated by carinal resection. Additional structures removed in these resections included a lobe of the thyroid, portions of the esophageal wall, and recurrent laryngeal nerve. Where extensive radiotherapy had been used prior to the operation, either remotely or recently, an omental pedicle flap was used to wrap the anastomosis (see Chapter 42, “The Omentum in Airway Surgery and Tracheal Reconstruction after Irradiation”). Gaissert and colleagues commenced review of the MGH experience with tracheal tumors to the year 2002.6 Figures cited here are therefore somewhat preliminary. In 40 years, 135 patients were treated for ACC and 135 for SCC. Overall resectability rates were 78% for ACC and 68% for SCC. Given that twice as many patients suffering from SCC were smokers, the significantly increased incidence of prior carcinoma of the lung (15%), larynx (7%), and other head and neck lesions (4%) is not surprising. The overall incidence of prior upper respiratory tract cancer in patients with SCC of the trachea was 27%. The principal reasons for nonresectability were extent of airway involvement (ACC 68%, SCC 67%) and extent of regional disease (ACC 23%, SCC 24%). Other causes were distant disease (ACC 6%, SCC 7%), medical contraindication (ACC 0, SCC 2%), and patient’s choice (ACC 3%, SCC 0).
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A
C
237
B
D
FIGURE 7-20 Very extensive adenoid cystic carcinoma which prohibited salvage of the larynx in a 39-year-old man. Diagnosis had failed to be made at an initial presentation with hoarseness and a paralyzed vocal cord 2 years earlier. Now he had dysphagia, dyspnea on effort, but no hemoptysis. The extent of laryngeal involvement precluded any attempt to salvage it. The patient was considered a candidate for cervicomediastinal exenteration (laryngotracheal pharyngoesophageal resection with mediastinal tracheostomy and esophageal replacement). Endoscopic views. A, Laryngoscopy. Tumor invades the subglottis up to the conus elasticus and fills the posterior commissure (arrow). B, Esophagoscopy shows submucosal tumor invading the esophagus anteriorly. C, Tomogram. Anteroposterior view. Tumor encircles the subglottis, paralyzes the vocal cord, and invades the upper trachea (open arrow). The solid arrow marks the glottis. D, Computed tomography delineation of tumor. Tumor infiltration of the larynx. The arch of thyroid cartilage is anterior, the remnant of cricoid posterior.
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F 7-20 (CONTINUED) E, Extension of tumor in the trachea, upper mediastinum, and around the esophagus. Carotid arteries are not invaded. F, Surgical specimen from another patient with adenoid cystic carcinoma, a 41-year-old man, viewed posteriorly. Tumor extends from the arytenoids downward. The esophagus was not involved. Total thyroidectomy, parathyroidectomy, and laryngotracheotomy were done. Because of the low level of mediastinal tracheostomy necessary, the brachiocephalic artery was divided and the omentum advanced. The patient later developed bone, lung, and liver metastases, which progressed slowly. He died from brain metastases 10 years later. FIGURE
E
Nearly 75% of patients undergoing tracheal resection for ACC and SCC were operated upon in the second two decades of this study (1982 to 2001), reflecting a variety of likely factors. The distribution of types of operation performed overall is listed in Table 7-7. The mean tumor lengths resected were 3.1 cm for all (standard deviation [SD] 1.7), 3.5 cm for ACC (SD 1.9), and 2.6 cm for SCC (SD 1.4).
Operative Results Complications that occurred in the series of 132 resections and reconstructions, which was comprised of consecutive experience from my first tracheal resections for tumors in 1962 until 1989, included the following: anastomotic stenosis in 2 after tracheal resection and in 4 after carinal resection; 3 air leaks; 4 suture line granulomas; 1 esophageal fistula after partial excision of the esophageal wall; unintended vocal cord paralysis in 8; and problems with aspiration or laryngeal function in 6, principally after laryngeal release. Stenoses were most likely due to excessive tension on the anastomosis, particularly following extended resection, abetted in one patient almost certainly by lengthy preoperative treatment with prednisone in high doses. All anastomotic stenoses were treated successfully by re-resection at a later date, although with a loss of reimplanted right upper lobe in 2 patients who had carinal reconstructions. Suture line granulomas antedated the use of absorbable suture material. Since absorbable Vicryl has been solely used for tracheal anastomosis, suture line granulomas have all but disappeared. Pulmonary edema occurred in 2 patients after right carinal pneumonectomy. Surgical complications are dealt with in detail in Chapter 21, “Complications of Tracheal Reconstruction.”
Primary Tracheal Neoplasms
Table 7-7 Types of Resection Performed For ACC and SCC of Trachea (2002) ACC Type of Resection Laryngotracheal Tracheal Tracheal with permanent tracheostomy Carinal without pulmonary resection Carinal with pulmonary resection Total
SCC
Total
n
%
n
%
n
%
8 45 7 23 18
8 45 7 23 18
8 57 5 18 2
9 63 6 20 2
16 102 12 41 20
8.4 53 6.3 21 10
101
100
90
100
191
100
Adapted from Gaissert HA et al.6 ACC = adenoid cystic carcinoma; SCC = squamous cell carcinoma.
In our 2002 review of all resections done over 40 years for ACC and SCC of the trachea, 4 of 101 patients with ACC and 7 of 90 with SCC required postoperative ventilation. Anastomotic separations occurred in 5 (5%) with ACC and 7 (8%) with SCC or 6.3% for both groups. Seven operative deaths in 132 patients undergoing tracheal and carinal resection and reconstruction totaled 5% (Table 7-8).1 One death (1%) occurred in the 82 patients undergoing tracheal resection; 6 deaths (12%) occurred among the 50 undergoing carinal reconstruction. The single death after tracheal reconstruction was due to anastomotic leakage and pneumonia. The deaths after carinal reconstruction were due to respiratory failure after anastomotic leakage, pulmonary edema, and pneumonia, and 1 from hemorrhage was probably due to pulmonary artery erosion. He was the single such patient in whom tissue had not been interposed between the anastomosis and pulmonary artery. Mortality was also high among the 9 patients who underwent exploration only, probably related to the extent of their malignant disease. As noted, failure and mortality rates in the group of staged reconstructions were high enough to discourage further use of this approach, except in extraordinary circ*mstances. Prostheses were not employed. The beneficial effect of growth of experience—patient selection, operative approach, improved techniques, increased comprehension of operative limits—is reflected in the progressive diminution of operative mortality for resection of ACC and SCC in successive decades: 21%, 11%, 5%, 3% (Table 7-9).
Oncologic Results In 1990, 135 of 147 patients were reported to have survived tumor resection.1 At the time of the survey, 49% of the patients with SCC, 75% of those with ACC, and 83% of patients with other tumors were alive and free of disease. These figures were difficult to interpret since the patients were spread over many years and accrued at a variable rate. The higher survival in the third group obviously reflected the large number of benign tumors and tumors of low malignancy in this group. In 2002, an analysis of the cumulative results of resection plus postoperative irradiation in nearly every case showed an absolute survival rate of 88% from operation for ACC; 86% at 1 year, 64% at 5 years, and 45% at 10 years.6 Treatment by irradiation only showed 44% survival at 5 years and 15% at 10 years (Table 7-10). Resection of SCC, with 95% survival of operation, gave survivals of 91% at 1 year, 46% at 5 years, and 25% at 10 years. Unresected survival was 8% at 5 years and 5% at 10 years (with irradiation) (see Table 7-10; Figure 7-21). A few unresected patients of both histologies underwent exploration only. The results cited for both types of cancers are for patients followed to the time of their death or within 18 months of enumeration (up to 40 years follow-up). In our earlier study, SCC of the trachea recurred in a pattern similar to that of squamous carcinoma of the lung; that is, within 5 years, and frequently with-
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Table 7-8 Results of Surgical Treatment of Primary Tracheal Tumors (1990) Variable Number of tumors resected Operative deaths (resection) Resection, reconstruction Trachea Carina Percent (resection reconstruction) Laryngotracheal Staged procedures Exploration only Survival (resected tumors) Dead Of tumor Of other cause Alive With tumor Without tumor Lost to follow-up
Squamous
Adenoid Cystic
Other
Total
44 3
60 8
43 1
147 12
1 1 5 0 1/2 2/6
0 4 8 0 4/6 1/3
0 1 2 0 0 0
1 6 5 0 5 3
13 6
7 5
3 1
23 12
0 20 2
1 39 0
0 35 3
1 94 5
Adapted from Grillo HC and Mathisen DJ.1
Table 7-9 Surgical Resection and Mortality Rates by Decade for Adenoid Cystic and Squamous Cell Carcinomas of Trachea and Carina Decade 1 2 3 4
Years
Number
Resection Rate (%)
Mortality Rate (%)
1962–1971 1972–1981 1982–1991 1992–2001
19 54 107 88
68 61 66 82
21 11 5 3
268
71
7
in 3 years (Table 7-11). Adenoid cystic carcinoma, on the other hand, recurred later, but continued to occur, sometimes many years later (see Table 7-11). Local recurrence, as well as metastases, have been observed 17 and 20 or more years after presumed complete resection. The slower rate of tumor progression and greater susceptibility to irradiation are probably both determining factors. As in most malignant neoplasms, a complete resection promises more favorable survival (Table 7-12). Five- and 10-year survival rates for ACC were 84% and 68%, respectively, with negative proximal and distal resection margins against 50%, and 28% with positive margins. Even more striking are the corresponding figures for squamous cell cancer of 52% and 30% versus 13% and 0% (Figure 7-22). Eschapasse collected 152 primary tumors from multiple teams in France and in the USSR.83 In 1974, he reported 121 patients treated surgically, 75 reconstructed after resection (47 trachea, 28 carina), with 13 deaths. Five of 19 patients with adenoid cystic tumors were alive and free of disease from 3 to 9 years, and 11 of 27 with SCC were alive and free of disease from 7 months to 16 years. In 1987, Perelman and
Primary Tracheal Neoplasms
Table 7-10 Absolute Survival from Adenoid Cystic or Squamous Cell Carcinoma of Trachea and Carina With and Without Surgical Resection Survival (%)
Adenoid Cystic Resected Unresected Squamous Resected Unresected Total
Number
Operation
1-year
5-year
10-year
81 30
88 97
86 86
64 44
45 15
87 41
95 95
91 68
46 8
25 5
239
93
84
84
26
Adapted from Gaissert HA et al.6 All patients were followed up to their death or within less than 18 months. Nearly all received postoperative irradiation, but dosage varied.
Koroleva reported 116 open operations on 153 patients with all types of tumors.85 Seventy-five underwent sleeve resection (n = 41) or carinal resection (n = 34), with 11 deaths. The actuarially calculated survival was 13% for SCC at 5 years and 66% for ACC at 5 years. Pearson and colleagues described 29 resections in 44 patients with primary tracheal tumors (16 sleeve resections, 13 carinal resections, 2 deaths).3 Nine of the patients with ACC were alive without disease from
FIGURE 7-21 Absolute survival of patients with adenoid cystic carcinoma (ACC) and squamous cell carcinoma (SCC), resected and unresected. Chart includes only patients with complete follow-up.
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Table 7-11 Survival after Resection of Tracheal Carcinoma (1990) Variable
Squamous
Adenoid Cystic
3
8
3(9)* 4(10) 3(6) 10(11)
5(11) 11(12) 10(11) 13(13)
Died of carcinoma > 10 yr 5–10 yr 3–5 yr 0–3 yr
0 0 5 8
4 1 2 0
Died without carcinoma; lost > 10 yr 5–10 yr 3–5 yr 0–3 yr
1 3 1 3
0 0 1 4
Alive with carcinoma
1
Operative deaths Alive without carcinoma > 10 yr 5–10 yr 3–5 yr 0–3 yr
Adapted from Grillo HC and Mathisen DJ.1 *Original number of operative survivors, excluding those who died of other causes later, is shown in parentheses.
1 to 20 years after operation. Three died of other disease 6 to 18 years postoperatively. Two were alive with disease. Four of 6 patients with squamous cell cancer were alive 6 to 56 months after resection. In 1996, Maziak and colleagues reported on 30 resections of 36 (83%) patients with ACC, with 7% mortality and survival rates of 79% and 51% at 5 and 10 years, respectively.11 Similar results were obtained in a smaller series.12 A collected series of 208 patients with primary tracheal tumors, from 26 institutions in France, Germany, and Italy, was composed of 94 with SCC, 65 with ACC, and 49 with other tumors.5 Of these
Table 7-12 Survival after Resection of Adenoid Cystic or Squamous Cell Carcinoma of Trachea and Carina With Negative or Positive Margins (2002) Survival (%) Number
Operation
1-year
5-year
10-year
Adenoid Cystic Negative margins Positive margins
30 49
87 88
90 83
84 50
68 28
Squamous Negative margins Positive margins
67 17
97 88
94 75
52 13
30 0
163
91
88
53
33
Total Adapted from Gaissert HA et al.6
Nearly all received postoperative irradiation of varied dosage. Margins are at proximal and distal resection lines. Lateral margins and lymph nodes are not noted here.
Primary Tracheal Neoplasms
FIGURE 7-22 Survival of patients resected for adenoid cystic carcinoma (ACC) and squamous cell carcinoma (SCC) with negative and positive margins. Margins are proximal and distal lines of resection.
patients, 165 underwent tracheal resection and primary anastomosis, 24 underwent carinal resection, and 19 underwent laryngotracheal resection, with an overall mortality of 10.5%. Survival at 5 and 10 years for ACC was 73% and 57%, and 47% and 36% for other tracheal cancers (squamous plus 4 with adenocarcinoma), respectively. Comparative observations on treatment of ACC by several groups are summarized in Table 7-13. The behavior of our remaining heterogeneous group of 48 tumors other than squamous cell or adenoid cystic carcinomas may be summarized as follows.1 Typical carcinoids did not recur, mucoepidermoid tumors behaved benignly in this series, and all benign tumors were cured. The few aggressive sarcomas generally suffered early recurrence. On the other hand, malignant fibrous histiocytoma, pseudosarcoma, and low-grade spindle cell sarcoma did not recur. Two small cell carcinomas, which had no evidence of nodal spread and were treated with additional standard chemotherapy, showed no recurrence in over 5 years. Unfortunately, these experiences are in anecdotal numbers only.
Radiotherapy Nearly all our patients with SCC, ACC, or sarcoma received postoperative irradiation. Radiotherapy was given since it is impossible to obtain sufficiently generous margins beyond the tumor in most tracheal resections because of the limited length of the trachea. In many cases, it was necessary to accept microscopically positive margins, especially with ACC, to permit reanastomosis. In some, adjacent lymph nodes were positive. Irradiation was given in an attempt to “sterilize” microscopic disease. The dose advised was in the range of 5,500 cGy (see Chapter 41, “Radiation Therapy in the Management of Tracheal Cancer”). The actual doses given varied since most patients were returned to their home communities for radiotherapy.
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Table 7-13 Treatment of Adenoid Cystic Carcinoma of Trachea Author (reference) Perelman (85)
Grillo (1)
Regnard (5)
Maziak (11)§
Gaissert (6)
Year of report
1987
1990
1996
1996
2002
Years included
20
26
23
32
40
Number of patients
—
80
—
38
135
Number of resected*
56
60
65
32
101
% resected
—
75
—
84
75
Operative mortality (%)
14†
13
6
9
11‡
Radiotherapy also (%)
41
100
43
81
100
Survival (%) 5 years
66
79
73
79
64
10 years
56
42
57
51
45
*Tracheal and carinal resections. †
All tracheal tumor resections.
‡
3% for 1992–2001.
§ACC
only
In our 1990 study, not unexpectedly, we observed that positive lymph nodes in SCC were more commonly present in patients who later died with cancer than in those who survived without cancer.1 Of 13 patients who died of cancer, 6 had positive lymph nodes. Of 22 disease-free survivors, only 2 had positive nodes. Invasive carcinoma at the resection margin had different consequences from in situ carcinoma. Four of 5 patients with invasive carcinoma died, whereas 6 with in situ carcinoma remained disease free. Parallel observations have been made in bronchogenic squamous carcinoma with respect to additional in situ lesions. Both lesions are sensitive to radiotherapy, in particular, ACC. Since it has been argued that radiotherapy might be considered as primary treatment for tracheal tumors, we examined the outcome of patients treated by resection followed by irradiation in comparison with those patients treated by irradiation alone (including patients explored but not resected).86,87 Although these are not totally comparable groups, the principal reason for not resecting the trachea in ACC was that the linear extent of the tumor was too great, rather than because of lateral bulk. In that sense, both shorter tumors and longer tumors may be considered somewhat comparable in accessibility to radiotherapy. Table 7-10 shows that there is a clear difference in survival of patients with both squamous and adenoid cystic tracheal carcinomas, when treated by resection followed by irradiation, when compared with irradiation alone (see Figure 7-21).1,6 Further, there appeared to be palliative benefit in giving irradiation after resection, even in those patients who ultimately died of recurrent carcinoma. The median survival of patients with SCC undergoing irradiation alone was 10 months, compared with 34 months for those who also were resected.1 Comparable figures for ACC are 28 months and 118 months, respectively. Cheung found primary radiotherapy for squamous cell and adenoid cystic carcinomas of the trachea in doses from 4,000 to 6,000 cGy to be ineffective in obtaining complete local control.88 Preoperative irradiation was briefly proposed by Pearson and colleagues but no data are available to support this use.3 The roles of external radiotherapy and brachytherapy are more fully expounded in Chapter 41, “Radiation Therapy in the Management of Tracheal Cancer.”
Recommendations for Treatment Based on our experience cited, and the experiences of other groups, we make the following recommendations for management of primary tumors of the trachea.
Primary Tracheal Neoplasms
1) 2) 3)
All benign primary tumors of the trachea and tumors of intermediate aggressiveness are best treated by complete surgical resection with primary reconstruction. Primary SCC and ACC of the trachea are best treated by resection followed by irradiation, as long as primary reconstruction is judged to be safely possible. Malignant primary tumors of other types should be resected if technically safe to do so, and irradiation most likely should be added despite small experience with many of these tumors.
The limited information that has accrued in the literature seems to support these recommendations.1,3–6,83,88,89 A final cautionary note is in order. During many years of treating tracheal tumors, I have seen numerous patients who have suffered prolonged delay in surgical management. Delay has first been due to the failure to make a timely diagnosis. The second significant cause of delay has been failure to offer definitive therapy because of lack of appreciation of the availability and effectiveness of current surgical techniques, assisted by irradiation. A third group of patients has been delayed for even less acceptable reasons: repetitive use of laser management without any clear rationale for continued use of this modality. These treatments were not given for emergency relief of obstruction, but rather as continuing management. Since the cure even of benign tracheal tumors is very rarely achieved by laser (or other endoscopic removal), such treatment is not justified, except perhaps for palliation of acute obstruction.90 Even then, there are simpler methods.91 Surgical resection, as early as possible, must be considered as the preferred initial treatment in almost every case.
References 1. 2. 3. 4. 5.
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Grillo HC, Mathisen DJ. Primary tracheal tumors. Treatment and results. Ann Thorac Surg 1990;49:69–77. Grillo HC. Primary tracheal tumours [editorial]. Thorax 1993;48:681–2. Pearson FG, Todd TRJ, Cooper JD. Experience with primary neoplasms of the trachea. J Thorac Cardiovasc Surg 1984;88:511–8. Perelman MI, Koroleva N. Surgery of the trachea. World J Surg 1980;4:583–91. Regnard JF, Fourquier P, Levasseur P. Results and prognostic factors in resections of primary tracheal tumors: a multicenter retrospective study. J Thorac Cardiovasc Surg 1996;111:808–14. Gaissert HA, Grillo HC, Shadmehr B, et al. Comparative long term survival after resection of adenoid cystic and squamous cell carcinoma of trachea and carina.[In preparation] Ranke EJ, Presley SS, Holinger PH. Tracheogenic carcinoma. JAMA 1962;182:519–22. Culp OS. Primary carcinoma of the trachea. J Thorac Surg 1938;7:471–87. Desai DP, Holinger LD, Gonzalez-Crussi F. Tracheal neoplasms in children. Ann Otol Rhinol Laryngol 1998;107:790–6. Saltarelli MG, Fleming MV, Wenig BM, et al. Primary basaloid squamous cell carcinoma of the trachea. Am J Clin Pathol 1995;104:594–8. Maziak DE, Todd TRJ, Keshavjee SH, et al. Adenoid cystic carcinoma of the airway: thirty-two year experience. J Thorac Cardiovasc Surg 1996;112:1522–32. Prommegger R, Salzer GM. Long term results of surgery for adenoidcystic carcinoma of the trachea and bronchi. Eur J Surg Oncol 1998;24:440–4. Soorae AS, Gibbon JRP. Primary oat-cell carcinoma of the trachea. Thorax 1979;34:130–1. Gelder CM, Hetzel MR. Primary tracheal tumours: a national survey. Thorax 1993;48:688–92.
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Ozturk S, Kutlay H, Sak SD, Yavuzer S. Fibrohistiocytic tumor of the trachea in a child. Eur J Cardiothorac Surg 1999;16:464–8. Sandstrom RE, Proppe KH, Trelstad RL. Fibrous histiocytoma of the trachea. J Clin Pathol 1978;70:429–33. Vinod SK, MacLeod CA, Barnes DJ, Fletcher J. Malignant fibrous histiocytoma of the trachea. Respirology 1999;4:271–4. Gonzalez-Campora R, Matilla A, Sanchez-Carrillo J, et al. ‘Benign’ fibrous histiocytoma of the trachea. J Laryngol Otol 1981;95:1287–92. Bando T, Fujimura M, Noda Y, et al. Pulmonary plasma cell granuloma improves with corticosteroid therapy. Chest 1994;105:1574–5. Mandelbaum I, Brashear RE, Hull MT. Surgical treatment and course of pulmonary pseudotumor (plasma cell granuloma). J Thorac Cardiovasc Surg 1981;82:77–82. Imperato JP, Folkman J, Sagerman RH, Cassidy JR. Treatment of plasma cell granuloma of the lung with radiation therapy. Cancer 1986;57:2127–9. Horowitz AW, Khalil KG, Verani RR, et al. Primary intratracheal neurilemoma. J Thorac Cardiovasc Surg 1983;85:313–20. Stack PS, Steckler RM. Tracheal neurilemmoma: case report and review of the literature. Head Neck 1990;12:436–9. Zeman MS. Carotid body tumor of the trachea. Ann Otol 1956;65:960–2. Choi G, Kim HY, Kim A, Choi Jo. Tracheal leiomyoma. J Otolaryngol 1998;27:87–9. Borski TG, Stucker FJ, Grafton WD, Nathan CO. Leiomyoma of the trachea: a case report and a novel surgical approach. Am J Otolaryngol 2000;21:119–21. Chen TF, Braidley PC, Shneerson JM, Wells FC. Obstructing tracheal lipoma: management of a rare tumor. Ann Thorac Surg 1990;49:137–9. Bateson EM. Relationship between intrapulmonary and endobronchial cartilage-containing tumors (so-called hamartomata). Thorax 1965;20:447–61. Butler C, Kleinerman J. Pulmonary hamartoma. Arch Pathol 1969;88:584–92. Logan PM, Miller RR, Muller NL. Solitary tracheal plasmacytoma. Can Assoc Radiol J 1995;46:125–6. Gomez-Roman JJ, Perez-Montes R, Perez-Exposito MA, et al. Primary lymphoplasmacytoid lymphoma of the trachea with immunoglobulin G paraprotein. Pathol Int 1999;49:1100–4. Fidias P, Wright C, Harris NL, et al. Primary tracheal nonHodgkin’s lymphoma. A case report and review of the literature. Cancer 1996;77:2332–8. Maeda M, Kotake Y, Monden Y, et al. Primary malignant lymphoma of the trachea. Report of a case successfully treated by primary end-to-end anastomosis after circumferential resection of the trachea. J Thorac Cardiovasc Surg 1981;81:835–9 Tse DG, Summers A, Sanger JR, Haasler GB. Surgical treatment of tracheomediastinal fistula from recurrent Hodgkin’s lymphoma. Ann Thorac Surg 1999;67:832–4. Grevend KM, Evans LS. The occurrence and management of esophageal fistulas resulting from Hodgkin’s disease. Cancer 1992;69:1031–3. Reichert K, Helling K, Mensen HD, et al. Primary tracheal leiomyosarcoma. J Laryngol Otol 2000;114:64–6. Postovsky S, Peleg H, Ben-Itzhak O, Arush MW. Fibrosarcoma of the trachea in a child: case report and review of the literature. Am J Otolaryngol 1999;20:332–5. Kegar A, Cantrel G, Rosen G. Rhabdomyosarcoma of the trachea. J Laryngol Otol 1988;102:735–6. Belda J, Canalis E, Gimferrer JM, et al. Subglottic stenosis in an HIV positive patient: an exceptional form of clinical presentation in Kaposi’s sarcoma. Eur J Cardiothorac Surg 1997;11:191–3.
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Zibrak JD, Silvestri RC, Costello P, et al. Bronchoscopic and radiologic features of Kaposi’s sarcoma involving the respiratory system. Chest 1986;90:476–9. Judson MA, Sahn SA. Endobronchial lesions in HIVinfected individuals. Chest 1994;105:1314–23. Weber AL, Grillo HC. Tracheal tumors. A radiological, clinical and pathological evaluation in 84 cases. Radiol Clin North Am 1978;16:227–46. Houston HE, Payne WS, Harrison EG Jr, Olsen AM. Primary cancers of the trachea. Arch Surg 1969;99:132–40. Hajdu SI, Huvos AC, Goodner JT, et al. Carcinoma of the trachea. Clinico-pathologic study of 41 cases. Cancer 1970;25:1448–56. Grillo HC. Tracheal tumors: surgical management. Ann Thorac Surg 1978;26:112–25. Eschapasse H. Les tumeurs trachéales primitives. Traitement chirurgicale. Rev Fr Malad Resp 1974;2:425–46. Valesky A, Hohlbach G, Schildberg FW. Wertigkeit unterschiedlicher massnahmen zur minderung der anastomosenspannung nach kontinuitätsresektion der trachea. Langenbecks Arch Chir 1983;360:59–69.
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Perelman MI, Koroleva NS. Primary tumors of the trachea. In: Grillo HC, Eschapasse H, editors. International trends in general thoracic surgery. Vol 2. Philadelphia: WB Saunders; 1987. p. 91–106. Rostom AY, Morgan RL. Results of treating primary tumours of the trachea by irradiation. Thorax 1978; 33:387–93. Makarewicz R, Mross M. Radiation therapy alone in the treatment of tumours of the trachea. Lung Cancer 1998;20:169–74. Cheung AYC. Radiotherapy for primary carcinoma of the trachea. Radiother Oncol 1989;14:279–85. Fields JN, Rigand G, Emami BN. Primary tumors of the trachea: results of radiation therapy. Cancer 1989; 63:2429–33. Daddi G, Puma F, Avenia N, et al. Resection with curative intent after endoscopic treatment of airway obstruction. Ann Thorac Surg 1998;65:203–7. Mathisen DJ, Grillo HC. Endoscopic relief of malignant airway obstruction. Ann Thorac Surg 1989; 48:469–75.
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CHAPTER EIGHT
Secondary Tracheal Neoplasms Hermes C. Grillo, MD
Thyroid Carcinoma Bronchogenic Carcinoma Other Tumors
Secondary neoplastic invasion of the trachea by direct extension is most often due to carcinomas of the esophagus, thyroid, and lung. Carcinoma of the larynx may also invade the trachea directly or it may recur at the margin of the stoma from lymphatics after laryngectomy. Less commonly, hematogenous metastases involve the trachea or carina. Sites of origin include the breast, melanoma, kidney, and thyroid. Carcinoma metastatic to the mucosa of the trachea from distant primary sites is less common than metastases to the bronchial mucosa, which is in itself an uncommon phenomenon. The goals of major resection of the trachea or carina for secondary neoplasms should be the possibility of cure, or otherwise, prolonged palliation. This excludes most hematogenous metastases. Palliation of irresectable obstructing tumor may be achieved by endobronchial curettage, laser therapy, external beam irradiation, brachytherapy, or sometimes by stenting. The limited place for tracheal resection and reconstruction, when the airway is invaded by adjacent neoplasm, is considered below. The two most appropriate categories for surgical treatment are thyroid and bronchogenic carcinomas. Invasion by esophageal carcinoma is almost never an indication for tracheal resection.
Thyroid Carcinoma Intraluminal airway invasion by differentiated thyroid carcinoma is rare, especially as a primary presentation, with estimates ranging from 0.5 to 7% or higher.1,2 Invasive well-differentiated thyroid carcinoma may present initially with hoarseness, hemoptysis, or dyspnea. Frequently, however, the invasive carcinoma is identified by a surgeon at thyroidectomy. The thyroid surgeon who finds the trachea invaded at the time of thyroidectomy commonly “shaves off ” the tumor from the tracheal wall. Treatment with 131I or external irradiation may follow. Unfortunately, such treatment may not be successful in preventing later airway obstruction. It has been calculated that 82% of deaths from thyroid carcinomas may be due to asphyxia or pneumonia from local recurrence.3–5 Invasion of the airway by differentiated thyroid carcinoma, either papillary or mixed follicular and papillary (which generally behaves like papillary carcinoma), is more common in older patients, but it is by no means restricted to this age group.
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Anaplastic or very poorly differentiated tumors frequently involve the trachea and larynx, to such degree at initial presentation that laryngeal salvage is not possible. The pharynx and esophagus may also be invaded in such advanced cases. Late, massive recurrence of differentiated carcinoma can produce a similar picture.
Tumor Behavior A well-differentiated thyroid carcinoma usually runs an indolent course, frequently with long-term survival.6,7 Airway invasion, however, is directly responsible for many late deaths due to thyroid cancer and is a source of profound morbidity from hemorrhage and suffocation.3 The cancer directly invades the closest portion of the airway. Tsumori and colleagues reported that 50% of papillary and follicular carcinomas which invaded the airway showed poor differentiation, whereas only 11.4% of noninvasive thyroid cancers of similar histology were poorly differentiated.8 Invasion also occurs most often in older patients, where papillary and follicular thyroid cancers may be more aggressive, although the spectrum is broad. Nomori and colleagues found that the nuclear area of tumor cells was significantly increased in cases with tracheal invasion compared to those without.9 Both groups noted that the tumors had sometimes become less well differentiated than at initial presentation.8,9 The prognosis of thyroid cancers invading the airway appears to correlate with the site and depth of invasion. Shin and colleagues classified papillary thyroid cancer invading the trachea as follows10 (Figure 8-1): Stage 0: Stage I: Stage II: Stage III: Stage IV:
tumor confined to the thyroid gland; extension through the capsule to abut the perichondrium but without cartilaginous erosion or intercartilaginous invasion; destruction of cartilage or intercartilaginous invasion; extension into the lamina propria of the tracheal mucosa; extension through the tracheal mucosa.
Clinical results correlated well with these stages, and in general confirmed the 1987 observations of Tsumori and colleagues.11 The manner in which papillary carcinoma invades the thyroid is by dissection along blood vessels and collagen fibers, oriented perpendicularly to the tracheal lumen between the cartilaginous rings.10 The perpendicular fibers course from the collagen fibers of the peritracheal fascia, which are parallel to the tracheal wall and are contiguous with those of the isthmus of the thyroid gland. Beneath the tracheal mucosa, these intercartilaginous fibers spread into a network of reticulum. Nerve fibers and lymphatics run parallel to the perpendicular collagenous fibers between cartilages. In a series of 22 patients with papillary cancer invading the trachea, these lymphatics rarely contained tumor. In the region of the isthmus, arteries emanate from the thyroid gland. The source of tracheal invasion is most often directly from the thyroid gland, along the anatomic pathways noted, and not primarily by lymphatic metastasis. Only en bloc excision will remove such invasion. Submucosal penetration indicates a poor prognosis. Because of the location of the thyroid gland, the subglottic larynx may also be invaded, most often in the cricoid cartilage (Figure 8-2). The corresponding recurrent laryngeal nerve is often paralyzed, paretic, or encircled by tumor. The adjacent esophagus and cricopharyngeus may be involved. The tumor may penetrate to any depth of the airway (Figure 8-3). Recurrent tumors are too often permitted to grow to large sizes before further surgery is contemplated, even though they may respond little to radioactive iodine uptake or to external radiotherapy. The added negative point of giving therapeutic radiotherapy where surgical reconstruction may later be necessary is obvious.
Diagnosis The patient with differentiated thyroid cancer involving the airway may present with classical symptoms and signs of airway neoplasm, namely, hemoptysis, wheezing, dyspnea on exertion, and, additionally, hoarseness.
Secondary Tracheal Neoplasms
FIGURE 8-1 Stages of papillary carcinoma of thyroid invading the trachea, based on the histopathologic extent of invasion. Adapted from Shin DH et al.10
More often, airway involvement produces no symptoms, since the tumor has not yet penetrated the mucous membrane or projected any distance into the lumen. A firm mass may be palpated, which is not freely movable over the trachea. Often, tracheal and laryngeal involvement are discovered at thyroidectomy. In my opinion, in addition to the usual diagnostic approach to thyroid cancer (thyroid function studies, thyroid scan, and needle biopsy), flexible bronchoscopy is advisable for every such patient, despite the rarity of visible airway invasion. However, there is not yet a completely accurate method to distinguish close abutment of the tumor to the tracheal wall from actual early invasion. Linear x-ray studies of the trachea include filtered views and crisp tomography, which are of great use in determining the extent of gross involvement of the larynx and trachea, and also the relative portion of the airway that is not involved (Figure 8-4). Fluoroscopy of the larynx adds information about the function of the vocal cords to that obtained by direct laryngoscopy (Figure 8-5). The neck should be imaged using thin section computed tomography (CT) scans, which are most likely to identify involvement of the tracheal wall or intrusion into the lumen (Figure 8-6). CT scanning should include the chest, to search for pulmonary metastases. Magnetic resonance imaging (MRI) is also useful in defining these lesions. Barium swallow may define the bulk of tumor and suggest esophageal involvement. Preoperative studies that are appropriate for exenteration, which may be considered for massive invasive tumors, are described in Chapter 34, “Cervicomediastinal Exenteration and Mediastinal Tracheostomy.”
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FIGURE
8-2 Papillary thyroid carcinoma in a 68-year-old male invading the upper trachea and left side of the cricoid.
Principles of Surgical Treatment The currently accepted principles of surgery for differentiated thyroid cancer call for complete removal of the local lesion and extensions in the neck. Longer survival and better control of symptoms are obtained if gross tumor is fully removed. This is usually interpreted to mean thyroidectomy and excision of involved regional lymph nodes, with persisting differences of opinion about the need for total thyroidectomy. Because of the pathological behavior of these tumors, nodal metastases are excised by limited regional dissection rather than by standard radical neck dissection. Even in extended node dissection, adjacent structures, such as the sternocleidomastoid muscle and internal jugular vein, are spared whenever possible. The submandibular triangle is rarely involved, but on the other hand, positive nodes do occur pretracheally, in the tracheoesophageal groove, along the length of the internal jugular chain, in the “V” between the innominate and left carotid arteries, and in the posterior cervical triangle. The goal of surgery, in addition to cure, is to prevent airway obstruction and death from asphyxiation. The otherwise guiding surgical principle of complete local removal of thyroid neoplasm is all too often broken when a tumor invades the upper trachea or the junction of the larynx and trachea. Frequently unfamiliar with techniques of airway surgery, the thyroid surgeon regards the addition of tracheal resection as “radical surgery,” potentially fraught with morbid or fatal consequences. Hence, “shave” techniques have been advocated.12–16 In experienced hands, however, airway reconstruction is not radical surgery. Addition of
Secondary Tracheal Neoplasms
A
B
FIGURE 8-3 A, Surgical specimen (mixed papillary and follicular carcinoma) of a patient, whose x-rays are shown in Figure 8-4, viewed from above. The broad arc of the partially resected cricoid is above in the photograph. Invading tumor is just below. B, The tumor extends between and through the cartilages. Seven years after resection of the obstructing tumor, the patient developed pulmonary metastases, which were treated with 131I.
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B
FIGURE 8-4 Radiologic delineation of linear extent of tracheal invasion by thyroid carcinoma. A, Filtered view of tracheal invasion by mixed papillary and follicular carcinoma in a 59-year-old man with identified pulmonary metastases. Arrow marks the glottis. Nonetheless, the patient enjoyed 14 years of life after tracheal resection and reconstruction, and had no further airway disease. B, Tomographic cut showing high invasion in the subglottic larynx by papillary carcinoma in a 36-year-old man. The right vocal cord is paralyzed.
tracheal resection following dissection for thyroidectomy adds little length or complexity to the operation and does not increase morbidity or mortality much.17,18 Voice, airway, and deglutition are all preserved. What is lacking are firm criteria about what constitutes an adequate “shave,” histologic identification of complete or incomplete tumor removal by shaving, the decades of follow-up necessary to validate this unusual oncologic approach, or consideration of the potential for change in the histology and aggressiveness of thyroid cancer.8,9 Many of our patients had been previously subjected to shaving procedures as initial and ultimately unsuccessful treatment, years before recurrence.17 Added to these considerations is the indication from our data that excision of airway involvement at initial thyroidectomy, or immediately thereafter, leads to better long-term results than late removal of a recurrent invasive tumor.17 The purposes of complete resection of thyroid cancer that invades the airway are, therefore, 1) to relieve or prevent airway obstruction in patients with slowly progressing neoplasm, 2) to prevent tortured death by asphyxiation or hemorrhage, and 3) perhaps to achieve cure by early complete resection of the tumor (Figure 8-7).
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8-5 Vocal cord paralysis or dysfunction due to recurrent laryngeal nerve invasion by papillary thyroid carcinoma, in a 36-year-old man, is well demonstrated on fluoroscopy or by direct examination. On these spot films taken during fluoroscopy, A, a paralyzed right cord is evident on attempted cord adduction. Note the asymmetry of the vocal cords. B, On inspiration, the tumor is also seen just below the glottis.
FIGURE
Radical removal of the larynx, trachea, and other affected tissues en bloc may be justified only in rare cases of seemingly confined undifferentiated carcinoma (Figure 8-8) and for palliation of longstanding massively recurrent and severely symptomatic differentiated carcinoma (Figure 8-9).17
A
B
FIGURE 8-6
Computed tomography scans of invasion by differentiated carcinoma. A, In a 65-year-old woman with papillary carcinoma invading the trachea, esophagus, cricoid, and right recurrent laryngeal nerve. Treated by thyroidectomy, with tracheal resection and reconstruction. B, In a 51-year-old man with a very large papillary lesion invading and compressing the trachea. Resection with reconstruction was nonetheless possible because the length of trachea invaded was less than the total extent of tumor apparent on the scan. Six years later, the patient remained free of recurrence.
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B A FIGURE 8-7
Severely obstructive papillary carcinoma in a 62-year-old man who had undergone thyroidectomy 6 years and 4 years earlier, followed by 131I. A, Tomogram shows marked occlusion of the proximal trachea (arrow). B, Resected specimen includes a portion of the cricoid cartilage.
A FIGURE 8-8 A, Computed tomography scan showing massive invasion of larynx, trachea, and esophagus by rapidly growing thyroid carcinoma of mixed Hürthle cells and anaplastic histopathology. This 71-year-old man was effectively palliated by cervicomediastinal exenteration for airway obstruction, with voice loss, total dysphagia and odynophagia, and head and neck pain. Mediastinal tracheostomy was established. With this histology, palliation was alone the goal. B, Gross surgical specimen of poorly differentiated squamous carcinoma of the thyroid in a 69-year-old man, similarly treated. He learned to use an electronic larynx well enough to continue to serve as town moderator. He died 6 years later of coronary disease, without recurrence of the thyroid cancer.
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8-9 A 62-year-old man with massive recurrent papillary thyroid carcinoma following thyroidectomy, 4 years previously, and subsequent treatment with 131I. The disease progressed to cause pain, bleeding, airway and esophageal obstruction, and loss of voice. The operative photograph shows the mass specimen including thyroid, larynx, trachea, and esophagus being removed by en bloc dissection. The neck is at the left. On the right, the upper sternum, heads of clavicles, and upper two costal cartilages have been excised to provide access for mediastinal tracheostomy. The flexible endotracheal tube in the right lower corner is in the proximal end of the trachea (arrow). The floor of dissection reveals carotid arteries, internal jugular veins, and prevertebral fascia. Good palliation was attained for a number of years. Pulmonary metastases appeared. FIGURE
Resection and reconstruction of the involved airway as part of a complete local excision of thyroid cancer, especially as an initial procedure, accomplishes the primary goals of conventional thyroid cancer surgery. It does not represent a radical extension of surgery attended by great hazards. Follow-up results strongly suggest that the best long-term results are obtained either 1) in those patients in whom the involved airway is removed at the initial resection of tumor or 2) where invaded airway is removed as soon as possible after identification at initial thyroidectomy. Prolonged palliation has been achieved by late removal of the airway invaded by a recurrent tumor, but it rarely provides a cure, even though patients had appeared earlier to run an indolent course. Palliative resection of an obstructed airway seems justified, even in the face of pulmonary metastases when the tumor is known to be slowly progressive.17 Pulmonary metastasectomy has not been shown to be of value in thyroid cancer.19 Most often, the recurrent laryngeal nerve that has to be sacrificed is already involved by tumor, and so no further functional loss follows.
Management Resection of the airway may require 1) simple circumferential removal of a segment of the upper trachea, 2) bevelled resection of one side of the anterolateral cricoid if it is involved, or 3) complex resection in which a portion of subglottic larynx on the invaded side is removed in a “bayonet” fashion and the distal trachea is tailored to repair the defect (Figure 8-10). “Window” resections are to be avoided because of the increased likelihood of leaving residual tumor and the less kindly healing of a trachea patched with
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FIGURE 8-10 Modes of resection of thyroid cancer invading tracheae. A, Cylindrical tracheal resection. Because of the location of the thyroid gland, invasion most frequently requires that proximal transection of the trachea be just below the cricoid cartilage. B, Varying amounts of cricoid must often be removed on the side of the tumor, from a slightly oblique bevelled resection to a nearly complete lateral excision, as diagrammed. C, “Bayonet” resection, where invasion of the cricoid is so extensive that the line of transection must lie somewhere beneath the vocal cord on that side. The inferior line of tracheal transection in this case is fashioned to fit the proximal laryngeal defect.
autologous mesenchymal tissue. The mesenchymal surface encourages granulation tissue formation and subsequent contraction. If such a window can be managed by insertion of a tracheostomy tube alone, the resection is usually of an inadequate extent. Surgical approach and techniques of resection and reconstruction are described in Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection,” and Chapter 25, “Laryngotracheal Reconstruction.” The technique of cervicomediastinal exenteration is described in Chapter 34, “Cervicomediastinal Exenteration and Mediastinal Tracheostomy.” Cervicomediastinal exenteration should be applied selectively; that is, only in the rare case where an invasive anaplastic or undifferentiated carcinoma appears to be totally resectable by such en bloc resection, or where there is highly symptomatic massive late recurrence of differentiated carcinoma, usually after multiple unsuccessful treatment by thyroidectomy, 131I, and sometimes external beam irradiation. Such an effort is principally palliative, but it does indeed provide the patient with a measure of comfort, as an alternative to the misery caused by a progressively extensive local disease.
Results of Treatment Total resection of the larynx and upper trachea was early performed for both well and poorly differentiated extensively invasive carcinoma of the thyroid, in some patients with surprisingly long-term palliation or apparent cure.20,21 Patients with a well-differentiated carcinoma involving the trachea or adjacent larynx, in limited enough fashion to be resectable along with the involved airway and yet permit primary reconstruction, were of course not completely resected prior to development of contemporary airway surgery. The techniques of airway reconstruction that evolved were only slowly applied in these patients. Grillo reported a case in 1965,22 whereas Ishihara and colleagues reported 11 patients in 1982.5 In 1986, Grillo and
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Zannini described 19 patients who underwent resection for papillary or mixed papillary and follicular carcinoma of the thyroid, and 3 patients for undifferentiated carcinoma.23 Sixteen of the patients had primary reconstruction performed and 6 were treated with en bloc cervicomediastinal resection with mediastinal tracheostomy. Fifteen of the 16 patients who underwent airway reconstruction had good surgical results and speech preservation. Eight patients were alive without disease up to 9 years later, and only 2 developed airway recurrence. In 1989, Maeda and colleagues recorded 151 patients with tracheoplasty for thyroid carcinoma in Japan, which represented nearly 27% of all tracheoplasties done in Japan up to that date.24 By 1991, Ishihara and colleagues had performed 60 resections for thyroid cancer, with 41 needing laryngotracheal anastomosis.18 Five- and 10-year survival rates were both at 78% with complete resection, and at 44 and 24% respectively with incomplete resection. In 1992, Grillo and colleagues described 34 of 52 patients who underwent resection, 27 with airway reconstruction and 7 with cervicomediastinal exenteration (Figure 8-11).17 Older patients predominated (Figure 8-12). Ten of the 27 reconstructed airways required laryngotracheal resections. The length of airway resection averaged 3.5 cm, a length that normally permits reconstruction without difficulty (Figure 8-13). Eighteen were not resected for reason of either distant metastases, excessive local extension, or to preserve laryngeal function where the only alternative was laryngectomy. In 2 of these latter patients, laryngotrachiectomy was deferred for a number of years. Of the 27 patients with reconstructed airways, it is notable that 9 had no prior treatment, 5 had been referred immediately after a surgeon identified invasion by tumor intraoperatively, and 13 arrived following recurrence at 1 to 47 years after the tumor had initially been shaved off the trachea. One patient died from local necrosis related to 4,800 cGy of irradiation, given 6 years previously, prior to the use of an omentum for vascular augmentation in previously irradiated tracheas (see Chapter 42, “The Omentum in Airway Surgery and Tracheal Reconstruction after Irradiation”). In a second patient, earlier postoperative tracheostomy would have avoided respiratory arrest due to edematous airway obstruction. The long natural history of differentiated thyroid carcinoma cautions on conclusions about longterm results (Figure 8-14). In the Massachusetts General Hospital (MGH) series, 11 of 25 patients died of cancer in 3 months to 10.25 years but only 2 had airway difficulty, indicating achievement of the principal goals—long-term palliation and obviation of death by airway obstruction.17 Twelve patients were without cancer for up to 14.5 years and 1 had pulmonary metastases. The average survival for 7 patients operated upon with known pulmonary metastases was 4.2 years, with the longest surviving at 10.5 years. Nine of 13 survivors without cancer had an airway resection done either as an initial treatment or were referred immediately after a surgeon discovered the invasion, which was far more promising than those treated for late recurrence after a remote prior thyroidectomy.
FIGURE 8-11 Treatment of patients with thyroid carcinoma invading the airway. Reproduced with permission from Grillo HC et al.17
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FIGURE 8-12 Distribution by age and gender of patients undergoing resection and reconstruction for thyroid carcinoma. Reproduced with permission from Grillo HC et al.17
8-13 Distribution of length of airway resected, where tracheal reconstruction was done. Reproduced with permission from Grillo HC et al.17 FIGURE
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FIGURE 8-14 Performance of 25 patients who survived tracheal resection and reconstruction for thyroid carcinoma. Reproduced with permission from Grillo HC et al.17
Recommendations The illustrative examples by Grillo and Ishihara and their colleagues17,18 have been amply confirmed by others in the last decade.17,18,25–30 Airway resection and reconstruction for differentiated thyroid cancer is safe, has a low morbidity and mortality and provides prolonged palliation, prevents death by asphyxiation or hemorrhage, and probably achieves cure in some patients. At issue with recommendation of the shave technique as a procedure of choice for stage I patients, is whether appropriate patients can be accurately selected by imprecise operative standards or by stratification of patients with presumed low-risk tumors, and the latter with certainty that the degree of differentiation will not change over the years. Also in question is the validity of surgical motivation to avoid simple extirpative surgery on the mistaken premise of high risk, where in fact added risk has been shown to be minimal and surgical results to be very good.12–16 Although this controversy will not be settled for some time, I support a consistent policy of complete local resection of all known tumor at the initial operation. Preoperative study to include CT and bronchoscopy will identify many patients who require airway resection and reconstruction. If the operator first discovers invasion at surgery and is uncomfortable with airway surgery, early referral for reoperation has proved to be effective.17 The larynx should be salvaged wherever possible, even if later removal might become necessary, even accepting an unknown increase in hazard of metastasis. 131I and especially external beam irradiation are better employed after thyroidectomy and airway reconstruction. Airway resection and reconstruction for late recurrent tumor in the airway provides the best palliation and prevention of bleeding and obstruction. In highly selected patients, radical resection including laryngectomy may be indicated.17,31
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Bronchogenic Carcinoma Bronchogenic carcinoma involving the proximal main bronchus (within 2 cm of the carina) is classified as a T3 lesion and that extending to the carina as T4. As surgical experience has grown, T3N0M0 lesions (including T3 lesions other than bronchial) have been moved to stage IIB, but T4N0M0 lesions remain in stage IIIB. The latter was based upon lack of familiarity with techniques of carinal resection and, in particular, of carinal pneumonectomy. A T4N0M0 lesion, by virtue of the carinal location, should be stage IIIA. T3 lesions due to main bronchial involvement, especially on the right, also usually require carinal resection in order to obtain an acceptable surgical margin. Squamous cell lesions centered at the carina may perhaps be considered as primary carinal neoplasms, in the way that a slightly more proximal lesion is a primary low tracheal carcinoma. The division is arbitrary but a localized central lesion is potentially treatable without loss of lung rather than by carinal pneumonectomy. Tumors suitable for excision by carinal pneumonectomy (or tracheal sleeve pneumonectomy) are predominantly squamous in type and right sided. Mathey and Jensik and their colleagues were among the earliest to practice sleeve pneumonectomy for bronchogenic carcinoma other than episodically.32,33 (For a fuller account, see Introduction, “Development of Tracheal Surgery: An Historical Review.”) Over a 15-year period, carinal resection for bronchogenic carcinoma was reported in North America by Deslauriers and colleagues, Jensik and colleagues, and Mathisen and Grillo, in Europe by Dartevelle and colleagues, Perelman and Koroleva, and Roviaro and colleagues, and in Japan by Isihara and colleagues and Watanabe and colleagues, among others.34–41 Excessive mortality initially accompanied this surgery but has improved with time (Table 8-1).
Governing Principles When bronchogenic carcinoma of the upper lobes invades the trachea directly from parenchymal contiguity, the disease is usually so extensive that segmental resection of the trachea is precluded. In a very rare circ*mstance, an extensive disease that invades the superior vena cava and adjacent edge of the trachea may still be grossly completely resectable. If the linear extent of tracheal invasion obviates segmental resection, lateral resection, despite its contraindications, may be considered. Repair may be made with a pedicled pericardium to preserve its viability, supported by Marlex. This repair must not be circumferential. Even so, granulations are likely to result and the hazard of mediastinal leakage is enhanced. A pedicled intercostal muscle may also be considered. On the other hand, when bronchogenic carcinoma is centered in the main bronchus or extends up to the carina, the patient should be considered for resection (see Chapter 29, “Carinal Reconstruction”). Carcinoma in the right upper lobe bronchus easily extends up the short length of the right main bronchus to the main carina. Carcinoma of the left upper lobe, with its considerably longer bronchus, is less likely to invade the carina. The number of right carinal pneumonectomies for bronchogenic carcinoma is therefore far greater than of the left. The disease must be localized enough so that resection of the
Table 8-1 Representative Results of Carinal Resection for Bronchogenic Carcinoma Author (Year) Jensik (1982) Deslauriers (1989) Watanabe (1990) Mathisen, Grillo (1991) Roviaro (1994) Dartevelle (1996) Mitchell (2001)
Reference
Operative Number
Mortality %
35 34 41 36 39 43 45
34 38 12 37 28 60 60
29 29 17 19 4 7 15
5-Year Survival % 15 13 19 20 43 42
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carina will not lead to reconstruction under tension. The usually safe limit for resection by right carinal pneumonectomy is approximately 4 cm in length between the points of the tracheal and left main bronchial division. Elevation of the left main bronchus is limited by the aortic arch. Carinal resection alone, with preservation of both lungs, can be somewhat more extensive because of the possibility for intrapericardial mobilization of the right lung, which is unrestricted by the aortic arch. In this case, the right main bronchus may be elevated in the thorax to reach the distal tracheal stump. The technique depends upon the length of tracheal resection. This also applies to the rarer left carinal pneumonectomy for bronchogenic carcinoma. In a few appropriate patients, the right lower or lower and middle lobes may be salvaged. Special precautions apply, as noted in Chapter 29, “Carinal Reconstruction.” Considerations regarding lymph node involvement are the same as for any resection of carcinoma of the lung. N3 disease conventionally places the disease out of bounds. N2 disease should be resected only as part of a protocol approach with neoadjuvant therapy, as in other cases of bronchogenic carcinoma. In these patients, the outlook remains poor. Because of the extent of the operation and a higher mortality rate, which is greater than for pneumonectomy alone, it is always important to search exhaustively for possible remote metastases prior to performing the resection. N2 disease heightens the probability of distant metastases. Carinal resection because of tumor invasion from metastases in subcarinal lymph nodes is not advised. In our series of 58 patients, 42 had squamous carcinoma, 10 had adenocarcinoma, 4 had large cell carcinoma, 1 had small cell carcinoma, and 1 had bronchoalveolar cell carcinoma.42 Particularly in right carinal pneumonectomy, significant interruption of tracheobronchial lymphatics necessarily follows, even where effort has been made to avoid a radical mediastinal lymphadenectomy.
Diagnosis and Evaluation Involvement of the carina by bronchogenic carcinoma must be assessed with great care by conventional imaging, which includes CT scan of the chest and upper abdomen. Crisp carinal tomograms can be useful to demonstrate the gross extent of the lesion, both within and without the lumen of the trachea, and to make clear the relative portion of airway that seems to be uninvolved by tumor (Figure 8-15). Final bronchoscopic assessment is best made with the Storz Hopkins magnifying telescopes through a rigid bronchoscope. Biopsies of tracheal mucosa proximal to the visible tumor may help to establish the feasibility of resection. Mediastinoscopy is very important for assessment of lymph nodes beyond the information obtained from CT scan. Mediastinoscopy is preferably performed concurrently with a planned resection so that tissue planes and definition of the tumor will not become obscured by inflammation and scar. Should preoperative adjunctive therapy be given following mediastinoscopy because of the finding of N2 lymph nodes, a further en bloc resection at a later date will have to encompass all node-bearing tissue, and accept partly obscured tissue planes. The role of positron emission tomography (PET) scanning in comparison with mediastinoscopy has yet to be clarified, but is likely to remain less exact. Metastases must be carefully sought by CT examination of the liver and brain and by bone scan. The increased surgical risk of the operation makes these studies mandatory. Patients being considered for carinal resection for bronchogenic carcinoma must be evaluated for total pulmonary function and distribution of ventilation and perfusion and blood gases. Smoking must have stopped. Sometimes, the involved lung contributes little to total respiratory function if the bronchus is severely obstructed. Cardiac function is also carefully assessed. In a few patients where pneumonectomy would not be tolerated, but where tumor is sufficiently localized, it is possible to perform carinal resection and right upper lobectomy with reimplantation of the middle and lower lobes or of the lower lobe (Figure 8-16). In such a case, involvement of the pulmonary artery is a crucial matter, and angiography may be needed. The final decision to proceed is made only after thorough exploration and before irrevocable surgical steps are
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B
8-15 A, Tomogram of large cell carcinoma (arrows) involving the lower trachea, carina, and right main bronchus in a 66-year-old man. B, Postoperative tomogram. The patient died 9 years later of other causes and without recurrence. FIGURE
taken. There are special hazards in such complex reconstruction that require precautions in order to avoid excessive anastomotic tension.
Management The technique for carinal resection (tracheal sleeve pneumonectomy) for bronchogenic carcinoma is described in Chapter 29, “Carinal Reconstruction,” and anesthesia in Chapter 18, “Anesthesia for Tracheal Surgery.” Resection of a carina and a bronchial stump for residual tumor or for strictly localized recurrence may be indicated, but only after rigorous preoperative and intraoperative assessment. Dartevelle and Macchiarini additionally recommend introducing latissimus dorsi and serratus anterior muscles to buttress the anastomosis, and performing a tailoring thoracoplasty to obliterate the pleural space for infection control.43 We have not done this in a “clean” operation. Initial experience with carinal resection for bronchogenic carcinoma was discouraging, with mortality rates of nearly 30% reported by Jensik, Deslauriers, and their teams.33,34 Our initial early postoperative mortality rate was 8%, but delayed mortality was 11%, due principally to anastomotic complications from tracheal reimplantation of the residual right lung, hence totalling a mortality rate of 19%.36 This contrasted with an 8 to 12% mortality rate for carinal resections for primary tracheal tumors.44 Nonetheless, inclusive of the unfavorable early cases noted, our total mortality rate dropped to 15.5% by 1998.42 The mortality rate of 11% reported by Dartevelle and colleagues in 1988 is assuring.37 Our surgical mortality dropped to 10% in the second half of our series, varying with type of resection.45 A very significant part of early postoperative mortality was due to a particularly aggressive and rapidly moving adult respiratory distress syndrome (ARDS), which has been labelled postpneumonectomy pulmonary edema or noncardiogenic pulmonary edema. The operation may go smoothly, and the patient who was extubated early will appear to be in fine
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FIGURE 8-16
A, Adenocarcinoma (arrows) of the right upper lobe invading the carina in a 40-year-old patient with insufficient pulmonary reserve to tolerate pneumonectomy. B, Reconstruction with end-to-side anastomosis of the bronchus intermedius to lower trachea (open arrow) just above the end-to-end joining of distal trachea and left main bronchus (arrow). Eight years later, an adrenal metastasis appeared. More often the bronchus intermedius is implanted into the medial side of the left main bronchus.
condition for 24 hours. At 36 to 48 hours, a diffuse infiltrate appears in the remaining lung. This progresses almost relentlessly to opacification of the residual left lung, and ultimately to death (see Figure 21-1 in Chapter 21, “Complications of Tracheal Reconstruction”). At postmortem examination, the lung is wet and heavy but only nonspecific bacteria, if any, are cultured. This does not support a postmortem diagnosis of bronchopneumonia. The syndrome also follows conventional right pneumonectomy less often, and left pneumonectomy or lobectomy even less frequently. Initially, it was attributed to perioperative intravenous fluid overload.46 We and others have not found any correlation between the amount of perioperative fluid administered and the occurrence of this dreaded complication.47 Nonetheless, it seems prudent to manage pneumonectomy patients with minimum fluid administration. Interference with pulmonary lymphatics may impair the ability of the remaining lung to clear interstitial fluid. Barotrauma is likely implicated. The low incidence of ARDS in the series of Dartevelle and Macchiarini suggested a difference in anesthesiologic techniques.43 The declining incidence of this complication is likely due to adjustment of intraoperative airway ventilatory pressure and tidal volumes to avoid pulmonary barotrauma. Until recently, the syndrome was nearly uniformly fatal. Addition of inhaled nitric oxide to the therapeutic regimen of fluid restriction, diuresis, ventilatory support, and steroids give promise of better results.47 Ten consecutive patients with severe ARDS (ARDS score 3.1), treated with inhaled nitric oxide at 10 to 20 ppm, showed immediate improvement in the mean ratio of partial pressure of arterial oxygen to fraction of inspired oxygen from 95 to 128 mm Hg (32% improvement), with further improvement there-
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after. Chest x-rays improved in 8 patients, and 7 patients survived. The 3 who died were late deaths related to sepsis after recovery from initial ARDS. Early in my experience, I tried to save the right lower or middle and lower lobes by bronchial implantation, after intrapericardial hilar mobilization, into either the side of the trachea above the end-to-end trachea to left main bronchus anastomosis, or into the medial side of the left main bronchus. Although the right main bronchus may be quite easily anastomosed in this way, excessive tension results if the right lower lobe bronchus or bronchus intermedius is pulled up to the trachea. Stenosis or separation resulted in a number of instances, with some fatal anastomotic complications.42 Clearly, this should not be done. If it is essential to save parenchyma because of borderline function, anastomosis of the bronchus intermedius or right lower lobe bronchus should be made to the medial side of the left main bronchus. Ishihara and colleagues also reattached the bronchus intermedius and left main bronchus to the trachea side-by-side, in 2 patients.40 Where functional status permits, pneumonectomy is probably preferable for safety. Unilateral node dissection does not seem to affect anastomotic healing. Excessive bronchial stripping is best avoided. As in all airway surgery in the thorax, a second tissue layer is advised over the anastomosis. If irradiation has been given remotely, omental coverage will help to provide healing elements to the inert bronchial tissues (see Chapter 42, “The Omentum in Airway Surgery and Tracheal Reconstruction after Irradiation”). Since these tumors are so central, resection will often be made intrapericardially, and portions of esophageal wall or superior vena cava may also have to be excised. Extensive surgery, including our earlier adverse techniques described, produced an operative morbidity of 47% compared with 27% for carinal reconstruction without pneumonectomy.42
Results At the outset, mortality rates from carinal pneumonectomy for bronchogenic carcinoma exceeded longterm survival rates (see Table 8-1). Jensik and colleagues reported a 5-year survival rate of 15% in 1982,35 wheras that from Deslauriers and colleagues was 13% in 1989,34 that by Dartevelle and colleagues was 23% in 1988,37 and that by Mathisen and Grillo was 19% in 1991.36 The figure of Dartevelle and Macchiarini rose to 43% in 1996.43 The overall survival rate from these authors and others was 22±12%. Dartevelle and Macchiarini found in 60 patients that long-term survival was significantly influenced by nodal status, with 5-year rates of 41, 54, and 0%, respectively, for N0, N1, and N2 lesions.43 Squamous cancer was more favorable than nonsquamous cancer. A 2001 review of the MGH series of resections of the carina by various surgical modes for bronchogenic carcinoma produced an overall 5-year survival rate of 42% (including operative mortality), with 19 absolute 5-year survivors.45 Survival was highest after isolated carinal resection (51%). Nodal involvement was a strong influence in this series as well. Five-year survival rates were 51% for N0, 32% for N1, and 12% for N2 or N3 lesions.
Comment With the development of carinal surgery, carinal pneumonectomy for bronchogenic carcinoma has become feasible, with growing safety. Involvement of the carina should not, therefore, in itself exclude consideration of surgery. Carinal resection is also appropriate to obtain an adequate margin for very proximal main bronchial carcinoma. Patients must be carefully appraised for the extent of local and distant disease and for the anatomic and functional feasibilities of safe resection. With careful attention to the selection of patients, surgical technique, and perioperative management, complications from the surgery should continue to decrease. Postpneumonectomy pulmonary edema or ARDS remains an ominous, if decreasing, threat. Its cause is not yet fully understood. Five-year survival rates of 30% or greater may be anticipated, if patients with N2 disease are excluded. Inclusion of N2 patients with neoadjuvant treatment will require careful prospective study.
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Other Tumors Recurrence of squamous cell carcinoma of the larynx at the tracheal stoma after laryngectomy has been approached by radical resection.48 This usually follows either postlaryngectomy irradiation, which failed to prevent recurrence, or failed irradiation treatment of recurrence. Resection frequently requires radical en bloc resection, which includes adjacent cervical tissues and, sometimes, the esophagus. Cervicomediastinal exenteration is described in Chapter 34, “Cervicomediastinal Exenteration and Mediastinal Tracheostomy.” Due to local recurrent tumor and prior irradiation, a wide excision may be necessary, with rotation of unirradiated myocutaneous flaps to cover the lower neck and upper mediastinum and with establishment of a low mediastinal tracheostomy. All too often, however, cancer recurs soon after resection. Paratracheal lymphatics are often permeated with cancer distally. Justification for such extensive surgery may therefore be questioned. For this reason, I have only performed a few cervical exenterations for recurrent laryngeal carcinoma. Cervical exenteration has also been performed for postcricoid squamous cell carcinoma of the esophagus, which involves the larynx and upper trachea, rendering laryngeal salvage impossible. Restoration of esophageal continuity may be accomplished by a variety of techniques.31 This, however, is beyond the scope of a book on airway reconstruction. Low cervical tracheostomy rather than mediastinal tracheostomy is usually possible in these patients. If abnormality is suggested, bronchoscopy and biopsy should be performed on all patients with upper and midesophageal carcinoma. Generally, segmental resection of the trachea, for direct invasion by esophageal carcinomas other than postcricoidal esophageal carcinoma, is not advised. The extent of involvement is usually so great that curative resection is unlikely. Tracheoesophageal fistula due to esophageal carcinoma is an extreme example. However, in a rare patient, following preoperative neoadjuvant treatment, if the only point of nonresectability is a short segment of trachea, it may be worthwhile to resect this segment and perform a direct tracheotracheal anastomosis. There is, however, a particular danger of tracheal necrosis, since the blood supply of the trachea is markedly diminished by adjacent esophagectomy. (See Chapter 1, “Anatomy of the Trachea,” describing tracheal blood supply.) Segmental arteries supplying anterior branches to the trachea and posterior to the esophagus are removed by esophagectomy. An undivided trachea retains viability via collateral vessels. However, when divided for anastomosis, the intramural blood supply may be insufficient for viability, and necrosis may occur adjacent to this anastomosis. To avoid complications, Matsubara and colleagues resected a limited portion of involved posterior tracheal wall and repaired the defect with a muscle flap.49 If combined resection is performed, advancement of the omentum is advisable. It is brought up with the stomach if this is used to reconstruct the esophagus, and separately if colon is used. Extension of a radical esophageal resection to include a tracheal segmental resection in any programmatic way is probably unwise. The palliation of malignant tracheoesophageal fistula, considered in Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula,” is not accomplished by tracheal resection. A unique case of esophageal leiomyomatosis involving the trachea has been recorded, necessitating esophagectomy and tracheal resection.50 Metastases from remote sites occur to the bronchi, but rarely to the trachea. King and Castleman found that over 185 of patients with pulmonary metastatic tumor also showed bronchial invasion by extension or metastatic deposit.51 Baumgartner and Mark described 2 endotracheal metastases from breast cancer, and cited 6 prior reports with primary sites in the breast, colon, kidney, and uterus.52 Heitmiller and colleagues found 1 tracheal metastasis in 23 patients with tracheobronchial metastases.53 Primary sites, in descending order of frequency, were the breast, kidney, and colon, with others from the bladder, thyroid, ovary, and nasopharynx. In most patients (87%), extrabronchial disease was also present. Although tracheobronchial metastases occurred late after primary tumor (mean 5 years), survival was short (mean 1 year).
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References 1. 2. 3. 4. 5. 6. 7. 8. 9. 10.
11.
12.
13. 14. 15.
16. 17. 18. 19. 20. 21. 22.
Lawson W, Som MP, Biller HF. Papillary carcinoma of the thyroid invading the upper air passages. Ann Otol Rhinol Laryngol 1977;86:751–5. Zannini P, Melloni G. Surgical management of thyroid cancer invading the trachea. Chest Surg Clin North Am 1996;6:777–90. Silliphant WM, Klinck GH, Levitin MS. Thyroid carcinoma and death: a clinicopathological study of 193 autopsies. Cancer 1964;17:513–25. Silverberg SG, Hutter RVP, Foote FW Jr. Fatal carcinoma of the thyroid: histology, metastases and causes of death. Cancer 1970;25:792–802. Ishihara T, Yamazaki S, Kobayashi K, et al. Resection of the trachea infiltrated by thyroid carcinoma. Ann Surg 1982;195:496–500. Cady B, Rossi R. An expanded view of risk-group definition in differentiated thyroid carcinoma. Surgery 1988;104:947–53. Grant CS, Hay ID, Gough IR, et al. Local recurrence in papillary thyroid carcinoma: is extent of surgical resection important? Surgery 1988;104:954–62. Tsumori T, Nakao K, Miyata M, et al. Clinicopathologic study of thyroid carcinoma infiltrating the trachea. Cancer 1985;56:2843–8. Nomori H, Kobayashi K, Ishihara T, et al. Thyroid carcinoma infiltrating the trachea: clinical, histologic, and morphometric analyses. J Surg Oncol 1990;44:78–83. Shin DH, Mark EJ, Suen HC, Grillo HC. Pathological staging of papillary carcinoma of the thyroid with airway invasion based upon the anatomic manner of extension to the trachea. Hum Pathol 1993;24:866–70. Tsumori T, Nakao K, Miyata M, et al. Clinicopathologic study on the mode and degree of invasion of the trachea by thyroid carcinoma. Nippon Geka Gakkai Zasshi 1987;88:600–6. Czaja JM, McCaffrey TV. The surgical management of laryngotracheal invasion of well-differentiated papillary thyroid carcinoma. Arch Otol Head Neck Surg 1997; 123:484–90. McCaffrey TV, Bergstralh EJ, Hay ID. Focally invasive papillary thyroid carcinoma: 1940–90. Head Neck 1994;16:165–72. Friedman M, Danielzadeh JA, Caldarelli DD. Treatment of patients with carcinoma of the thyroid invading the airway. Arch Otol Head Neck Surg 1994;120:1377–81. Nishida T, Nakao K, Hamaji M. Differentiated thyroid carcinoma with airway extension: indication for tracheal resection based on the extent of cancer invasion. J Thorac Cardiovasc Surg 1997;114:84–92. McCarty TM, Kuhn JA, Williams WL, et al. Surgical management of thyroid cancer invading the airway. Ann Surg Oncol 1997;4:403–8. Grillo HC, Suen HC, Mathisen DJ, Wain JC. Resectional management of thyroid carcinoma invading the airway. Ann Thorac Surg 1992;54:3–10. Ishihara T, Kobayashi K, Kikuchi K, et al. Surgical treatment of advanced thyroid carcinoma invading the trachea. J Thorac Cardiovasc Surg 1991;102:717–20. Protopapas AD, Nicholson AG, Vini L, et al. Thoracic metastasectomy in thyroid malignancies. Ann Thorac Surg 2001;72:1906–8. Frazell EL, Foote FW Jr. Papillary cancer of the thyroid: a review of 25 years of experience. Cancer 1958;11: 895–922. Hendrick JW. An extended operation for thyroid carcinoma. Surg Gynecol Obstet 1963;116:183–8. Grillo HC. Circumferential resection and reconstruction of the mediastinal and cervical trachea. Ann Surg 1965;162:374–88.
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Grillo HC, Zannini P. Resectional management of airway invasion by thyroid carcinoma. Ann Thorac Surg 1986;42:287–98. Maeda M, Nakamoto K, Ohtea M, et al. Statistical survey of tracheobronchoplasty in Japan. J Thorac Cardiovasc Surg 1989;97:402–14. Mellière DJM, Ben Yahia NE, Becquemin JP, et al. Thyroid carcinoma with tracheal or esophageal involvement: limited or maximal surgery. Surgery 1993;113: 166–72. Ozaki O, Sugino K, Miura T, Ito K. Surgery for patients with thyroid carcinoma invading the trachea: circumferential sleeve resection followed by end-to-end anastomosis. Surgery 1995;117:268–71. Bayles SW, Kingdom TT, Carlson GW. Management of thyroid carcinoma invading the aerodigestive tract. Laryngoscope 1998;108:1402–7. Musholt TJ, Musholt PB, Behrend M, et al. Invasive differentiated thyroid carcinoma: tracheal resection and reconstruction procedures in the hands of the endocrine surgeon. Surgery 1999;126:1078–87. Park CS, Suh KW, Min JS. Cartilage-shaving procedure for the control of tracheal cartilage invasion by thyroid carcinoma. Head Neck 1993;15:289–91. Kim KH, Sung MW, Chang KH, Kang BS. Therapeutic dilemmas in the management of thyroid cancer with laryngotracheal involvement. Otol Head Neck Surg 2000;122:763–7. Grillo HC, Mathisen DJ. Cervical exenteration. Ann Thorac Surg 1990;49:401–9. Mathey J, Binet JP, Galey JJ, et al. Tracheal and tracheobronchial resections. Technique and results in 20 cases. J Thorac Cardiovasc Surg 1966;51:1–13. Jensik RJ, Faber P, Milloy FJ, Goldin MD. Tracheal sleeve pneumonectomy for advanced carcinoma of the lung. Surg Gynecol Obstet 1972;134:231–6. Deslauriers J, Beaulieu M, McClish A. Tracheal sleeve pneumonectomy. In: Shields TW, editor. General thoracic surgery. 3rd ed. Philadelphia: Lea and Febinger;1989. p. 382. Jensik RJ, Faber LP, Kittle CF, et al. Survival in patients undergoing tracheal sleeve pneumonecotmy for bronchogenic carcinoma. J Cardiovasc Thorac Surg 1982;84:489–96. Mathisen DJ, Grillo HC. Carinal resection for bronchogenic carcinoma. J Thorac Cardiovasc Surg 1991; 102:16–23. Dartevelle PG, Khalife J, Chapelier A, et al. Tracheal sleeve pneumonectomy for bronchogenic carcinoma. Report of 55 cases. Ann Thorac Surg 1988;46:68–72. Perelman M, Koroleva N. Surgery of the trachea. World J Surg 1980;4:583–91. Roviaro GC, Varoli F, Rebuffat C, et al. Tracheal sleeve pneumonectomy for bronchogenic carcinoma. J Thorac Cardiovasc Surg 1994;107:13–8. Ishihara T, Ikeda T, Inoue H, f*ckai S. Resection of cancer of lungs and carina. J Thorac Cardiovasc Surg 1977; 73:936–43. Watanabe Y, Shimizu J, Oda M, et al. Results in 104 patients undergoing bronchoplastic procedures for bronchial lesions. Ann Thorac Surg 1990;50:607–14. Mitchell JD, Mathisen DJ, Wright CD, et al. Clinical experience with carinal resection. J Thorac Cardiovasc Surg 1999;117:39–53. Dartevelle P, Macchiarini P. Carinal pneumonectomy for bronchogenic carcinoma. Sem Thorac Cardiovasc Surg 1996;8:414–25. Grillo HC, Mathisen DJ. Primary tracheal tumors: treatment and results. Ann Thorac Surg 1990;49:69–77.
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Mitchell JD, Mathisen DJ, Wright CD, et al. Resection for bronchogenic carcinoma involving the carina: longterm results and effect of nodal status on outcome. J Thorac Cardiovasc Surg 2001;121:465–71. Zeldin RA, Normandin ED, Landtwing BD, Peters RM. Postpneumonectomy pulmonary edema. J Thorac Cardiovasc Surg 1984;87:359–65. Mathisen DJ, Kuo EY, Hahn C, et al. Inhaled nitric oxide for adult respiratory distress syndrome after pulmonary resection. Ann Thorac Surg 1998;65:1894–902. Krespi YP, Wurster CF, Sisson GA. Immediate reconstruction after total laryngopharyngoesophagectomy and mediastinal dissection. Laryngoscope 1985;95:156–61. Matsubara T, Ueda M, Nakajima T, et al. Can esophagec-
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tomy cure cancer of the thoracic esophagus involving the major airways? Ann Thorac Surg 1995;59:173–7. Kumar P, Breach NM, Goldstraw P. Esophageal leiomyomatosis involving trachea: surgical resection and repair. Ann Thorac Surg 1997;63:531–3. King DS, Castleman B. Bronchial involvement in metastatic pulmonary malignancy. J Thorac Cardiovasc Surg 1943; 12:305–15. Baumgartner WA, Mark JB. Metastatic malignancies from distant sites to the tracheobronchial tree. J Thorac Cardiovasc Surg 1980;79:499–503. Heitmiller RF, Marasco WJ, Hruhan RH, Marsh BR. Endobronchial metastasis. J Thorac Cardiovasc Surg 1993;106:537–42.
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CHAPTER NINE
Tracheal and Bronchial Trauma Hermes C. Grillo, MD
Mechanical Injuries Burns
Mechanical Injuries Injuries to the trachea and bronchi, whether blunt or penetrating, are uncommon. Figures for frequency depend upon the numerator (type and location of injury, venue) and the denominator (autopsy series, multiple trauma cases, survivors).1
Types of Injuries Airway trauma may be life threatening, immediately or in the hours following acute injury. Penetrating or blunt trauma to the neck may injure the larynx or cervical trachea, whereas injuries to the thorax may damage the thoracic trachea and bronchi. In civilian practice, blunt trauma to the airway from motor vehicle accidents used to be the most common cause of such injuries. Unfortunately, penetrating wounds from stabbing and gunshot have increased rapidly in urban areas, with firearms in the lead (Table 9-1).1 Cervical injuries also result from striking wires or cables while driving motorcycles or snowmobiles, or from strangulation by seat belt.2,3 In motor vehicle injuries, the larynx and cervical trachea are damaged by direct impact of the steering wheel or dashboard against the cervical vertebra, often with the neck extended. Direct blows to the neck also produce laryngotracheal injuries. Blunt trauma to the anterior thorax during motor vehicle accidents, or less often from industrial crush injuries, produces a variety of injuries to the thoracic trachea, carina, and main bronchi. Penetrating injuries to the trachea are most often due to bullet or stab wounds and may be accompanied by major vascular trauma. Because of the lethal nature of missile or stab wounds to the surrounding major structures, penetrating wounds of the thoracic trachea are not often seen in the living. The esophagus is at risk in all of these injuries, cervical and thoracic. Tracheobronchial lacerations, which result from endotracheal intubation or other per oral instrumentation, are included here. Postintubation injuries, which are late results of treatment of respiratory failure, are described in Chapter 11, “Postintubation Stenosis.”
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Table 9-1 Type of Injury and Location Blunt Cervical airway Thoracic trachea Bronchi
28% 27% 45%
65 62 105
100%
232
Penetrating 29% 63% 90%
77% 17% 6%
161 36 12
100%
209
Total 71% 37% 10%
226 (100%) 98 (100%) 117 (100%) 441
Reproduced with permission from Bacha EA et al.1
Cervical Injury. Damage to the larynx and cervical trachea from blunt injury may include fracture with or without displacement of the hyoid, the supraglottic larynx, the infraglottic larynx, the cricoid cartilage, and the cervical trachea (Figure 9-1). A variable amount of trachea may be elevated into the neck by cervical hyperextension at the time of injury, depending on the age of the patient. In the young, the larynx may be severely contused or otherwise injured without actual fracture of the more flexible cartilages. In older patients, stiffening and calcification render the larynx more vulnerable to fracture. Mucosal tears and avulsions are seen. Vocal cords and arytenoids may be torn and displaced. It is important to consider the spectrum of possible injuries when examining an acutely traumatized patient.4 Separation of the airway may be partial or complete. The points of actual rupture of the cervical airway are most commonly between the cricoid and trachea and in the upper trachea. Blunt trauma rarely produces clean cuts, but rather produces complexes of injuries, including, for example, cricotracheal separation with concurrent fracture of the cricoid cartilage, and avulsion of mucosa from the anterior surface of the posterior cricoid plate (Figure 9-2). In 19 patients with laryngotracheal disruption, 11 due to direct impact and 8 due to strangulation, Couraud and colleagues identified 14 complete separations below the cricoid or first ring.3 Nine cricoid fractures were seen. The mucosa retracted in all patients to expose cricoid cartilage. One or both recurrent laryngeal nerves may be temporarily or permanently damaged. Fourteen of Couraud’s patients suffered bilateral recurrent nerve damage and 4 were unilateral. Concomitant tears of the esophagus may occur. Avulsion of the trachea from the cricoid may be accompanied by transverse laceration of the anterior esophagus from the pharynx, where it is attached to the cricoid posteriorly, or by completely circumferential separation. Subluxation of cervical vertebrae, with or without injury to the spinal cord, may occur concomitantly. With penetrating wounds of the neck and thoracic inlet, and even with blunt trauma, the spectrum of potential injuries includes major vascular injuries. Penetrating cervical wounds, chiefly due to stab or gunshot wounds, may injure the trachea as one of several structures damaged. Bilateral recurrent laryngeal nerve division is less common than in complete tracheal separation due to blunt injury. Although dissenting voices are raised, surgical exploration of penetrating cervical wounds still seems to be the judicious course. Thoracic Trachea and Bronchi. Fracture or laceration of the thoracic trachea, carina, or main bronchi following closed chest injury may be seen in children and young adults without rib or sternal fractures. The young thorax can absorb major compressive trauma and rebound without skeletal fractures. In the older patient, clavicular and upper rib fractures and the number of rib fractures correlate with the likelihood of tracheobronchial injury.5 It is likely that a sudden increase of intrabronchial pressure against the closed glottis, or rapid deceleration, produces some airway injuries. Blunt injuries to the thoracic trachea are most common in the lower trachea and vary widely from complete transection to a partial horizontal tear. Vertical splitting of the trachea from the carina upward occurs, running anteriorly through cartilages and/or posteriorly up the membranous wall, either in its center or laterally along the junction with the cartilages. Injury to the lower trachea may be accompanied by partial or complete shearing of one or both main bronchi.
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A
B
C
D
E
FIGURE 9-1 Cervical laryngotracheal blunt trauma. A, Supraglottic tears and fractures. B, Transglottic injuries. C, Cricoid fracture. D, Avulsion of trachea from cricoid. E, Laceration or tear of trachea. Adapted from Harris HH. Management of injuries to the larynx and trachea. Laryngoscope 1972;82:1924–9.
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FIGURE 9-2 Blunt injury to the neck in motor vehicle accident. The distal end of the trachea is completely separated from the larynx. The forceps holds a posterior and lateral full thickness mucosal flap (arrow) which was avulsed from the anterior surface of the posterior cricoid below the arytenoids. The endotracheal tube (ET) has been introduced through a tracheostomy below the level of transection. Since both recurrent laryngeal nerves were severed, it was evident that a tracheostomy would be necessary postoperatively and hence the ET was placed through this location. The mucosal flap was resutured into the posterolateral laryngeal defect. A laryngeal mold was also used. The esophagus, which had been separated from the pharynx, was reanastomosed. Strap muscle was interposed between laryngotracheal and pharyngoesophageal suture lines.
Lobar or segmental bronchi may also be lacerated or separated by crush injuries, usually accompanied by deep parenchymal laceration. From a review of 183 tracheobronchial blunt injuries reported between 1970 and 1990, Symbas and colleagues noted that 74% were transverse ruptures with 4% in cervical and 12% in thoracic trachea, 25% in the right main bronchus, 17% in the left main bronchus, and 16% in lobar bronchi (Figure 9-3).6 Of longitudinal tears (18%), 6.5% were in the cervical trachea, 10% in the thoracic trachea, and 1.5% in main bronchi. The 8% remaining were complex, involving the trachea and right or both main bronchi. Most injuries occur within 2.5 cm of the carina. Kiser and colleagues, in a review of 265 patients who suffered blunt tracheobronchial injuries, confirmed a greater frequency of right-sided injuries, both overall and at the time of diagnosis and treatment.7 They found that bronchial rupture occurred within 2 cm proximal to the carina in 76%, and that 43% occurred in the right main bronchus. Recurrent laryngeal nerve injury is rare in thoracic tracheal trauma. Also rare but equally as important is concurrent laceration of the esophagus, often longitudinally. Esophageal injury should be considered in every posterior laceration of the trachea, since esophageal injury from blunt trauma is unlikely to occur by itself. It probably results from sudden forceful compression of the trachea and esophagus against the vertebrae, such as that from steering wheel impact. The injury occurs more often in young patients, with or without upper rib fractures. An elastic chest wall seems to favor such injury. Injury is most common in the lower trachea but may occur in the neck. The communication may be instantly established or occur later as traumatized tissues necrose. Potentially lethal mediastinitis may be a consequence of an overlooked intrathoracic esophageal laceration. Failure to recognize acute injury to the airway because of distraction due to catastrophic associated injuries, or failure to manage acute airway injury appropriately, may lead to cicatricial obstruction and other sequelae, days or months later. Such problems are often correctable but with greater difficulty and complications. Tracheobronchial Lacerations after Intubation. Lacerations of the trachea and bronchi may be produced by single and double lumen endotracheal tubes. The injuries are in the membranous wall and are usually
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linear. Postoperative mediastinal or subcutaneous emphysema may indicate an unrecognized intraoperative endotracheal intubation laceration. In a careful analysis of these lacerations, Massard and colleagues found predominant injuries in the lower trachea and main bronchi, and in the case of single lumen tubes, along the right membranous cartilaginous junction.8 Cervical laceration occurs less often. Overinflation of cuffs rather than stylets or tube tips appears to cause the injuries. Repositioning a tube that was originally placed in the right main bronchus without deflating the cuff may be an important factor. Short women, with correspondingly narrower airways, appear to be more at risk. Lacerations also occur from the placement of tracheostomy tubes when insertion is difficult. Mediastinal or subcutaneous emphysema and pneumothorax are harbingers. The modalities of surgical repair and conservative treatment are discussed in Chapter 31, “Repair of Tracheobronchial Trauma.” In general, small lacerations may be safely managed conservatively, but larger ones are best repaired surgically.8,9 Injury to the membranous wall of the trachea above the carina or of the left main bronchus has been noted in about 1 to 2% of patients undergoing transhiatal esophagectomy.10 These have been treated by suture, reinforcement with pleura or pericardium, and effective buttressing with the gastric tube neoesophagus.
9-3 Blunt tracheobronchial injuries: type and location. Adapted from Symbas PN et al.6 RMB = right main bronchus.
FIGURE
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Clinical Characteristics Acute Injury. Cervical tracheal injury presents most commonly with palpable subcutaneous emphysema. Contusion, abrasion, or laceration of skin may or may not be present. Hemoptysis is a common finding and hoarseness, inspiratory stridor, and dysphonia or aphonia may occur. The patient may be in acute respiratory distress with asphyxia or show little initial difficulty.11,12 Slight respiratory distress which is present initially may suddenly precipitate into severe obstruction. Subcutaneous emphysema may increase with coughing or swallowing. Deep cervicomediastinal emphysema is characteristic and pneumothorax may be present.2,3 Laceration of the thoracic trachea, the main bronchi, or both may produce dyspnea, subcutaneous emphysema, massive cervical and mediastinal emphysema, or uni- or bilateral pneumothorax. Pneumo mediastinum or cervical emphysema is a warning of probable tracheal rupture. Bilateral pneumothorax often indicates tracheal or carinal rupture. Unilateral or tension pneumothorax may occur, particularly with bronchial rupture. Hemopneumothorax may be present. Mild hemoptysis occurs. A mediastinal “crunch” may be heard, synchronous with heartbeat (Hamman’s sign). Response to chest tube suction varies. There may be partial resolution of pneumothorax or none, continued brisk air leak, and even increased dyspnea on suction. These responses should alert the surgeon to a probable airway rupture. On the other hand, the main bronchus may be torn without pneumothorax, especially if there is preexisting pleural obliteration. These tears are more often in the lower trachea or carina. If the esophagus is also torn, it may not immediately be manifest clinically.13 The earliest sign of esophageal injury may be cough on swallowing, becoming noticeable in the days following injury, or the onset of mediastinitis. Concurrent injuries to organs in the traumatized region must therefore be sought and treated at the outset. These may include injury to the aorta, brachiocephalic and/or carotid arteries. Thirteen of 15 patients with early and late blunt tracheobronchial trauma at all levels had the following associated injuries: rib fractures (10), esophageal injuries (7), pulmonary contusion (5), vascular injury (4), head injury (4), and extremity fractures (4).14 Chronic Injury. In 3 of 17 patients, whom we saw for late management of upper tracheal trauma, diagnosis had not been made at the time of injury.2 In 10 of these patients, tracheostomy had been performed to establish a secure airway but no attempt at primary repair had been made. In these patients, complete airway stenosis had usually evolved above the tracheostomy at the level of the injury, and in addition, vocal cord paralysis was present. In 4 cases, primary repair had been attempted but failed, resulting in airway stenosis. Among the 3 patients whose diagnosis was overlooked, progressive airway obstruction developed between 1 to 4 weeks after injury. One patient also developed a tracheoesophageal fistula, with such acute surrounding inflammation that proximal esophageal defunctioning, a procedure we rarely use, was required before correction could be attempted. The absence of pneumothorax following injury or its apparently successful management by intrapleural suction may lead to failure to diagnose intrathoracic tracheal or bronchial injury. The area of separation heals by cicatrization, causing tracheal obstruction or bronchial stenosis with atelectasis or obstructive lung infection.
Diagnosis In many patients with acute injury to the trachea, and to the cervical trachea in particular, clinical signs will strongly suggest the diagnosis. However, tracheobronchial injuries should be considered in every patient with severe trauma to the neck or chest in order to avoid overlooking any injuries. Bronchoscopy should be performed whenever the possibility of injury is suspected. With cervical injuries, anteroposterior and lateral radiographs of soft tissues of the neck as well as chest radiographs may reveal dissection of air in the cervical tissues up to the base of the skull or into the mediastinum (Figure 9-4). Mediastinal emphysema,
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B
A 9-4 Blunt tracheal fracture. The esophagus was also avulsed from the pharynx. A, Lateral cervical roentgenogram. Note dissection of air throughout cervical tissues to base of skull. Oral contrast has diffused into the tissues. B, Computed tomography (CT) scan reveals air throughout cervical tissue planes at the level of thyroid and cricoid cartilages. C, CT section at the level of tracheal transection shows air anteriorly, with extravasated contrast behind. FIGURE
C
with cervical dissection of air and subcutaneous emphysema, is usually present in intrathoracic tracheal rupture. Pneumothorax, sometimes bilateral, will often be present unless there is pleural obliteration. Complete bronchial rupture may reveal the lung collapsed at the bottom of the thorax, when only the pulmonary vessels retain continuity. Partial but significant collapse, unresponsive or poorly responsive to suction, may present when bronchial mediastinal pleural investments are partially intact. At other times, the lung will fully expand on suction, sometimes without subsequent air leak, which may be clinically deceptive. Pleural obliteration may limit even mediastinal emphysema (Figure 9-5). Laryngoscopy is necessary to assess possible damage in the uppermost airway. This is facilitated by the flexible nasopharyngoscope. In acute injury, accurate laryngeal assessment may be very difficult because of edema, hemorrhage, and tissue trauma. If the first manifestation of the injury is airway obstruction, initial treatment also serves as a diagnostic procedure. Bronchoscopy should be done early for direct visual-
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A
B
C
D
FIGURE 9-5
Variations in radiographic picture obtained following main bronchial rupture. A, Complete right main bronchial rupture with lung collapsed at the base of hemithorax, since it remains connected to the hilum only by pulmonary vessels. Tension is indicated by mediastinal displacement. Hemothorax is present. B, Partial expansion of lung on pleural suction after right main bronchial fracture, because peribronchial tissues maintain a partially “intact” air channel to the lung. This presentation is most commonly seen. Note the mediastinal emphysema and air in the muscle planes. C, Chest roentgenogram of a 69-year-old woman, run over by a truck. Multiple bilateral rib fractures and unstable chest wall. Mediastinal shift to right. Pleural obliteration prevented pneumothorax. D, Same patient after partial atelectatic collapse of left lung. Air delineates the transected left main bronchus.
ization of the airway. Flexible bronchoscopy with an endotracheal tube threaded over it may be the method of choice (see Chapter 31, “Repair of Tracheobronchial Trauma”, and Figure 10-1 in Chapter 10, “Tracheostomy: Uses, Varieties, Complications”). Pneumothorax may be severe or accompanied by tension and must be treated immediately.
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If there is suspicion or any possibility of cervical spine injury, urgent airway assessment and establishment must be done with this concern in mind. Adequate splinting must be provided during radiographic examination. Intubation over a flexible bronchoscope avoids cervical flexion or extension. If that fails, urgent tracheostomy becomes necessary. This is one of the few remaining indications for emergency tracheostomy. Equipment for tracheostomy should be at hand when bronchoscopy is first attempted, in case of loss of airway during endoscopy. In a series of acute tracheal injuries, only 4 patients needed no intubation, whereas 11 required oral endotracheal intubation—2 with the aid of a flexible bronchoscope, 2 intubated through the open neck, and 2 with a rigid bronchoscope.14 Eight airways were controlled by tracheostomy. Computed tomography (CT) adds detail but is usually not critical to diagnosis, and it certainly should not be routinely obtained if the patient is unstable or might lose the airway (Figure 9-6). In an urgent situation, sufficient information is usually at hand after endoscopy and on the x-rays to proceed to surgical treatment, unless there is strong indication for additional imaging such as angiography. If esophageal injury is suspected, contrast esophagography is performed after the airway is secure, and other life-threatening injuries, such as aortic rupture, have been evaluated. A small amount of barium or Gastrografin is used. Contrast studies may fail to show esophageal injury. Therefore, rigid esophagoscopy is performed if cervical spine injury is absent. The proximity of a penetrating injury is sufficient to raise the question of esophageal injury.13 If the injury on the initial chest x-ray suggests the possibility of vascular injury, then a CT scan with contrast or, more definitively, angiography is advised to delineate the lesion precisely (Figure 9-7). In injuries where diagnosis has been delayed or only an emergency tracheostomy was established, the larynx is first completely and carefully assessed. This is best done by an experienced otolaryngologist on the awake patient, so that glottic function is fully observed. Complete radiographic studies of the larynx and trachea are performed (see Chapter 4, “Imaging the Larynx and Trachea”). If indicated, esophagography is included (Figure 9-8). Endoscopic examination is next, made of all portions of the airway and of the esophagus if necessary. In many patients in whom treatment is delayed, cicatrization produces total discontinuity between the larynx or upper trachea and the distal trachea. Access to the lower airway is via tracheostomy only (Figure 9-9). Frequently, there will appear to be a long gap between the two ends of the airway. Since the distal end of a separated trachea drops into the mediastinum, whereas the upper segment remains fixed to the less
FIGURE 9-6 Computed tomography scan at and just below the carina showing a separated left main bronchus following blunt chest trauma (arrow). Massive emphysema is seen in all layers of the chest wall.
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B
9-7 Blunt injury to the neck caused separation of the cervical trachea with recurrent laryngeal nerve injury and damage to the right carotid artery resulting in aneurysm, demonstrated in A, anteroposterior and B, lateral arteriograms. The injury occurred 2 weeks earlier. FIGURE
mobile larynx, the apparent major airway gap is usually illusory. In 18 patients seen, late repair was occasioned by failure of initial diagnosis in 9, failed repairs in 8, and due to tracheoesophageal fistula and respiratory failure in 1.14 The importance of correct laryngeal assessment cannot be overemphasized. It is sometimes necessary that corrective procedures be applied to the damaged or paralyzed larynx prior to reconnection to the distal trachea (see Chapter 35, “Laryngologic Problems Related to Tracheal Surgery”). If the tracheal separation were corrected first, and only then was it discovered that the airway at laryngeal level was inadequate, the patient would require immediate reintubation through the larynx or a tracheostomy. This could adversely affect the integrity of the tracheal repair. The proper order of business, therefore, is restoration of the larynx to anatomical and functional adequacy, prior to repair of the trachea or sometimes concurrently with airway restoration. Management of acute and chronic airway trauma is described in Chapter 31, “Repair of Tracheobronchial Trauma.” If early repair of tracheal separation is not done, it is best to wait 4 to 6 months before delayed repair is performed so that inflammation may subside and scar may mature, providing that obstruction is not present.
Results Larynx and upper trachea. If principles of management are strictly observed for isolated laryngeal and upper tracheal trauma (see Chapter 31, “Repair of Tracheobronchial Trauma”), the results of treatment of acute injuries are generally very good (Table 9-2). Since tissues beyond the actual area of injury are essentially
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normal, precise reconstruction with accepted techniques generally produces a permanently satisfactory airway. In a series of 10 patients treated promptly for acute injuries, all achieved an excellent airway, and there were no instances of later tracheal stenosis.2 Couraud and associates treated 19 patients with laryngotracheal disruption; 11 within 5 days of injury, 17 by similar repairs but with stenting in 13, and 2 by laser and stenting.3 Excellent respiratory results were achieved in all. Phonation was good in 7 of the patients and fair in 13, reflecting the impossibility of restoring true vocal cord normalcy. Schaeffer reported excellent results in treatment of acute laryngeal trauma.4 Early definitive treatment was emphasized based on classification by severity of the laryngeal injury, with observation only in the absence of mucosal laceration or cartilaginous fracture and displacement. Functional results are generally better, following early rather than delayed repair. Delayed management of these injuries presents a multitude of problems, depending on the individual injury and the nature of the prior treatment. Where both recurrent laryngeal nerves are permanently damaged, a wholly adequate albeit husky voice can be obtained. A paralyzed larynx can still function satisfactorily for speech. The glottic aperture must be fixed at approximately 4 mm (see Chapter 35, “Laryngologic Problems Related to Tracheal Surgery”). This provides for clear speech, which is produced largely by the pharyngeal musculature, with the lung bellows providing an air column in a more efficient way than the stomach and esophagus do for so-called “esophageal speech” after laryngectomy. Modulation of voice is lacking. A high school senior who had suffered tracheal separation with bilateral cord paralysis reported, after repair of glottis and trachea, that he was able to return to the debating team but not to the glee club. If the glottic aperture is made narrower, speech may be improved, but the patient will not be able to move
A
B
FIGURE 9-8 Late findings 6 months after a motorcycle-cable injury in a 15-year-old, resulting in high tracheal separation and esophageal avulsion at the cricopharyngeus. The left vocal cord was paralyzed and the right functioned suboptimally. A, Anteroposterior tomographic section showing severe stenosis of cervical trachea. The subglottic larynx lies at the top. Arrow at site of stricture. B, Lateral neck view with swallow of barium. Arrow indicates stricture of the trachea. Severe stenosis of the esophagus is demonstrated by contrast medium. Both injuries were successfully corrected surgically in a single procedure.
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FIGURE 9-9 Another 15-year-old who suffered a motorcycle-cable injury 4 months earlier. Airway obstruction was emergently treated with tracheostomy at an outside hospital, and subluxation of C2-C3 was managed with Crutchfield tongs and traction. After initial recovery, the patient was able to swallow liquids and semisolids only. A, Contrast outlines the subglottic larynx and a short segment of the proximal trachea. The arrow marks the point of transection. The channel below is the esophagus. B, Lateral view shows barium in the larynx above (at left) passing through a narrow fistula (arrow) to the distal esophagus. Retracted pouch of avulsed pharynx is seen behind the larynx on the right with a blind sinus below it. Tracheostomy provided access to the distal trachea. C, Lateral view following reconstruction of trachea and esophagus. Air column of trachea is seen (at left) anterior to the contrast-filled upper esophagus, showing a good lumen at the site of repair. Thyrohyoid muscle was interposed between the suture lines. The right vocal cord had sufficient function to provide satisfactory voice. Swallowing returned satisfactorily.
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an adequate amount of air and will be short of breath on exercise. If the aperture is much larger, the patient will be able to exercise more intensely but will lose adequacy of speech and be more susceptible to aspiration. Dysphonia is the most important late disability after laryngotracheal injury.12
Tracheal and Bronchial Trauma
Table 9-2 Outcome of Airway Injuries
Blunt Cervical Thoracic trachea Bronchial Total Penetrating Cervical Thoracic trachea Bronchial Total Reproduced with permission from Bacha EA et al.
Injuries
Deaths
Mortality (%)
16 14 32
0 2 8
0 14 25
62
10
16
151 31 11
11 11 3
7 35 27
193
25
13
1
In 17 patients on whom delayed reconstruction of the airway was done, 16 attained a good airway without limitation of activities.2 One patient preferred to maintain a tracheal cannula so that he could open it intermittently during strenuous exercise. Variable results were obtained with vocal function. A good voice was attained in 10 patients, 3 of whom had unilateral and 4 of whom had bilateral vocal cord paralysis. Six patients had a husky but functional voice. Three of these had unilateral paralyses and 3 had bilateral paralyses. In 1 case, adequate vocal function was not achieved. This patient had one paralyzed cord and another with partial paresis. He was able to speak only in a whisper. No patient with tracheoesophageal fistula repaired by the technique described (see Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula”) had subsequent recurrence of the fistula. Thoracic Trachea and Bronchi. High mortality is often associated with intrathoracic tracheal and bronchial trauma but this is due to severe associated injuries or widespread body trauma (see Table 9-2).11 Blunt injuries of sufficient force to damage the tracheobronchial tree are likely to injure other organs. Penetrating wounds easily injure major structures centered in the thorax. Good anatomic and hence functional results can now be expected following tracheobronchial repair of acute injuries, whether linear or anastomotic,6,12 if performed in accordance with current techniques. Poorly conceived or executed reconstructions are likely to fail. Late tracheal injuries, usually presenting as a stenosis, are successfully managed by resection and reconstruction (Figure 9-10).2,14 Main bronchial stenosis from overlooked injuries (Figure 9-11) or failed repair should be managed by careful dissection and reanastomosis, even if the lung has been functionless for a long time. It appears that the proportional return of function is roughly inversely proportional to the length of time elapsed. Deslauriers and colleagues demonstrated a quite respectable return of lung function after reanastomosis.15 If chronic sepsis or fibrosis has supervened, pneumonectomy is indicated to remove a septic source. Lesser treatments, including dilation, laser, and steroid injection, have no lasting benefit. Generally, repair of fresh injuries is accomplished most often by suture repair, with minimal debridement, and resection is rarely required. Late repairs most often need resection with reconstruction.14 In 28 patients with early and late repairs, at all levels, only 3 suffered airway complications; 1 early dehiscence was promptly repaired and 2 late separations were treated with T tubes, one of which remained permanently.14 Death from liver failure occurred in 1 cirrhotic patient. Individual patients present special challenges, both with respect to type and location of injury, concomitant trauma, and prior treatment and timing (Figure 9-12).6
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Burns Inhalation Burns Burns of the larynx, trachea, and bronchi may be caused by inhalation of hot gases, steam, particulate matter in smoke, or of chemical substances released in industrial explosions or combustion. We have encountered burns due to house fires, motor vehicle accidents, electrical exposure, gas explosion, airplane crash, TV set explosion, and chemical inhalation (ammonia and hydrochloric acid), among many other origins of burns.16 It is often difficult to know precisely what the mixture of damaging agents was. In addition to heat, injury may be produced by irritant gases such as aldehydes, ammonia, and hydrochloric acid, and by particulate matter. Moylan and Chan observed by bronchoscopy that one-third of burn patients had evidence of inhalation injury.17 Ninety-seven percent of these had facial burns and 75% were injured in a
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FIGURE 9-10 Findings in a patient who developed progressively severe dyspnea late after anterior chest trauma. A, Bronchoscopic examination shows tight, well-healed stenosis in midtrachea. B, Gross appearance of the site of left-sided transverse laceration of mediastinal trachea (arrow). The wedge defect brings to mind a woodsman’s attack on a tree trunk. C, Resected specimen of stenosis. The stenotic airway is about one-fourth or less of the normal tracheal cross section. Also, see Figures 21 and 22 (Color Plate 14).
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B 9-11 A 26-year-old patient suffered multiple trauma in a motor vehicle accident 5 months earlier, with initial bilateral pneumothoraces which responded to tube drainage. With late onset of dyspnea, a right main bronchial stenosis with nearly complete occlusion was identified. Two attempts were made at different institutions to dilate the stenosis. A, Inspiratory chest x-ray shows apparently good bilateral expansion. B, Expiratory film shows continued full expansion on the right due to air trapping. C, Bronchoscopy reveals nearly complete occlusion of the right main bronchus at the origin. Obstruction is not complete, however. Resection of the stenosis and reanastomosis of the right main bronchus allowed her to return to climbing in the Grand Tetons. Also, see Figure 23 (Color Plate 14). FIGURE
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closed space. Seventy-five percent of these patients developed severe respiratory complications and onethird of them died as a result of airway burns. The patient often also suffers from cutaneous burns of varying extent and depth. When first examined, the patient may have significant burns of the oropharynx, largely of thermal origin. These commonly heal and regress so that the glottis may soon appear quite normal. Varying degrees of persisting injury are observed in the subglottic larynx and upper trachea, the worst being just beneath the glottis with gradual diminution of the effects of the burn proceeding distally.16 These burns appear to result more from chemicals and particulates in smoke, except in the case of steam burns. An exaggerated necrotizing process
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FIGURE 9-12 Example of complex tracheobronchial injury in an 8-year-old girl, thrown from a vehicle and crushed as it rolled over. A, Initial chest roentgenogram. Bilateral pneumothoraces (intubated) and extensive emphysematous dissection of chest wall muscles and planes. There is little evidence of rib fractures or displacement, due to flexibility of the rib cage in childhood. At the initial hospital, laparotomy was negative. Tracheal transection was identified and presumptive repair was done under cardiopulmonary bypass. B, Subsequent chest film shows complete opacification of the left chest. Eight days after injury, she was urgently transferred. The initial repair was found to be inadvertent anastomosis of trachea to right main bronchus rather than to trachea. The concurrent rupture of the left main bronchus went unrecognized. At right thoracotomy, the tracheobronchial repair and right hilum were mobilized intrapericardially. The left bronchial stump could not be freed under the aortic arch because of inflammation and early scar formation. This required mobilization via left thoracotomy. Left intrapericardial mobilization was also done and the aortic arch was freed. Finally, via sternotomy and transpericardial dissection between the vena cava and aorta, and after proximal tracheal mobilization, it was possible to implant the debrided left main bronchus in the left lateral wall of trachea above the prior anastomosis. A second anesthesia machine was used to reinflate the left lung prior to anastomosis, vastly improving oxygenation. If the pathologic findings after 8 days could have been anticipated, alternative incisions might have been possible. C, Tomogram showing final reconstruction. The upper arrow marks the glottic level. In the neocarina, the right main bronchus appears long due to deviation of the distal trachea to the right, below the left bronchial anastomosis (lower arrow). The patient’s further course was excellent.
immediately below the cords was first noted in victims of The Cocoanut Grove fire and was attributed to eddy currents. Some unfortunate patients sustain injury extending into the main bronchi and below. The intensity of damage varies. Severe tracheobronchitis may be followed by mucosal sloughing. If the basal cell
Tracheal and Bronchial Trauma
layer remains intact, early repair is accomplished rapidly, both clinically and experimentally. If the basal membrane is destroyed, granulations, cicatrization, and stenoses may follow. A late complication of inhalation burn, 2 to 6 months after acute injury, is the formation of endobronchial polyposis.18,19 Significant hemoptysis occurs. Polyps regressed over 6 months without treatment in one patient and while receiving corticosteroids in another. Fatal bronchiolitis obliterans has been described as a late complication of inhalation burn following explosion in a confined space.20 Effects of inhalation injury and the intubation injury resulting during treatment are difficult to separate, especially since intubation is often performed early in the presence of respiratory symptoms. Burn injury may well make the trachea more susceptible to intubation injury. Stenoses are often of greater length than those resulting from intubation injury alone. Injury may also extend beyond the level of grossly visible changes. Often, the cartilages of the trachea appear to be only slightly injured. Peritracheal fibrosis is almost always found. The incidence of stenosis due to burn injury is impossible to determine, given the variety and intensity of agents and the widespread use of intubation in management. Clinical reports are largely composed of patients who were intubated. Two of 38 survivors of burns treated with intubation developed subglottic stenosis in one series, and in another, 6 of 25 survivors of airway complications treated with tracheostomy developed tracheal stenosis. In a search for later sequelae of inhalation burns in 17 survivors, 4 had tracheal stenosis and 5 had significant tracheal granulomas. Gaissert and colleagues treated 18 patients with chronic airway compromise after inhalation burns; there were 18 tracheal stenoses, 14 subglottic strictures, and 2 main bronchial stenoses.16 Three patients developed laryngotracheal stenoses without intubation. Evaluations of patients with inhalation burns include tracheal and laryngeal radiography followed by laryngoscopy and bronchoscopy under general anesthesia. In our 18 chronic patients, 14 had subglottic as well as tracheal stenoses, and in 4, the two areas were separated by a tracheal segment which was not stenosed. If airway obstruction occurs, whether by laryngeal edema, inflammatory swelling, granulations, or later stenosis of the burned subglottic larynx and trachea, an airway is best established urgently by endotracheal intubation. Obstruction may also occur 3 weeks to 5 months after injury. If the patient has pulmonary damage that requires ventilation, a cuffed tube is necessary. Otherwise, it is preferable to avoid an inflated cuff. For long-term management, tracheostomy becomes necessary if the patient’s neck is not damaged by the burn. The tracheostomy usually lies within the damaged area of the trachea. As inflammation subsides, a T tube may be inserted to span the entire area of injury (see Chapter 39, “Tracheal T Tubes”). If the subglottic larynx is involved, as it often is, the T tube must extend up through the glottis. This is frequently necessary because the intensity of an inhalation burn is often greatest in the subglottic larynx. A T tube, with its upper end between the false and true vocal cords, usually permits hoarse or whispered speech as well as swallowing without aspiration. Training by a speech pathologist is advisable. If the proximal end of the T tube is sited in the subglottic location where there is burn injury, it will repeatedly become obstructed by granulation tissue. The T tube maintains airway patency, preserves understandable phonation, and permits gradual resolution of burn injury to the mucosa and submucosa. Burn injury resolves only very slowly, and resulting cicatrization matures slowly, which is entirely parallel with the evolution of cutaneous burns. Attempts to perform surgical resection and early reconstruction of the airway are likely to fail.16 With patience and persistence by both the patient and surgeon, conservative management is most likely to result in a satisfactory airway, although not a normal one (Figure 9-13). Since cartilages remain basically intact, the goal is regression of granulations and stabilization of the mucosal and submucosal process. In 5 patients treated with T tubes only, and 4 with laryngofissure and T tube, decannulation was achieved between 4 to 61 months (mean 28 months) after injury. Four patients required permanent tracheal tubes (2 T tubes and 2 tracheostomy tubes). Specific criteria for discontinuance of the T tube based on bronchoscopic observation or biopsy do not exist. Our management has been to attempt to remove the T tube when regression seemed adequate,
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B
A
C
9-13 Lateral cervical roentgenograms from an 18-year-old female suffering inhalation burn by toxic gases from combustion of plastic building materials. A, Diffuse upper tracheal stenosis (arrow). Posterior laryngeal calcification is visible superiorly. B, T tube in place, extending from the subglottic larynx into normal trachea. The sidearm of the T lies at the base of the neck. C, Result after 6 years of splinting is an apparently stable although narrowed subglottic larynx and proximal trachea. Dense reactive scar tissue appeared to have resolved to a degree. However, the patient went on slowly to laryngotracheal stenosis, which was successfully treated by laryngotracheal resection and reconstruction 13 years after decannulation (C). She has required several procedures for posterior commissural glottic stenosis and unilateral arytenoid fixation over subsequent years. The laryngotracheal repair has remained stable. FIGURE
leaving a cannula or “button” in place to maintain the stoma during the test period. If resection of a limited residual stenosis is necessary, it is preferably done later than earlier, although good response has been reported to earlier repair on occasion. Operative management is even more hazardous because so many burns involve the subglottic space, where airway reconstruction is more difficult, whether by single-stage laryngotracheal reconstruction or by laryngofissure with resurfacing and stenting. Four of 6 patients who ultimately underwent open repair of a subglottic stenosis had good results. Management of stenosis extending into the carina and main bronchi, fortunately rare, is even more difficult. We hesitate to use a T-Y tube since the bronchial ends of the tube may stimulate more reaction if they lie in areas of burn injury. Isolated bronchial stenosis has been managed by repeated dilation. An inlying stent would pose the danger of inciting granulomas. Successful outcome of treatment does not result in a normal airway. The quality of voice is often diminished and a degree of hoarseness is present. Mild chronic wheezing and recurrent episodes of respiratory tract infections occur. We have seen a case of late recurrent obstruction, but there is no large experience with these injuries.
Ingestion Burns Ingestion of caustic substances such as lye may produce burns of the oropharynx, larynx, and esophagus. The epiglottis may be destroyed, and the vocal cords injured severely enough to fibrose. Tracheal ingestion is not common, perhaps due to the protective reflexes of the glottis. Severe caustic injury to the upper esophagus may on rare occasion penetrate by necrosis through the back wall of the trachea or left main bronchus. Tracheal injury is usually distal.
Tracheal and Bronchial Trauma
Attempts at conservative management of such injuries penetrating from the esophagus have not succeeded. Appropriate treatment, although not sufficiently documented, would seem to be removal of the destroyed esophagus, conservative debridement of the airway injury, and patching with appropriate tissue. A carefully sutured intercostal muscle pedicle flap, using multiple fine sutures placed as if in an anastomosis and not simply as “tacking” sutures, should provide protection. The omentum should also be considered for buttressing in such a catastrophic situation.
Laser Burns Despite warnings and precautions, occasionally, a plastic endotracheal tube has been ignited by a laser, producing disastrous tracheal and bronchial thermal burns.21 Such a lesion is managed in the same fashion as inhalation burns, but the extent and depth may well be fatal, especially if the injury extends into the bronchial tree. Even Y or T-Y stents become useless. Similar burns have been produced by inept use of the cautery during tracheostomy. A laser burn of the left main bronchus was incurred in an ill-advised and ill-executed attempt to destroy an area of dysplasia. The resulting complete stenosis of the bronchus was successfully treated by bronchial resection and anastomosis (see Figure 38 [Color Plate 16]). A tracheoesophageal fistula produced by laser treatment of cuff stenosis was managed by standard reconstructive techniques. Since the laser damage was localized, reconstruction was feasible in this case. Injuries caused by external irradiation are discussed in Chapter 41, “Radiation Therapy in the Management of Tracheal Cancer.” I have encountered an irremediable main bronchial stenosis due to a misguided use of brachytherapy to irradiate peribronchial tissue rather than the intralumenal tumor. The resulting complete stenosis was fused to the adjacent pulmonary vessels.
References 1. 2. 3. 4. 5. 6. 7. 8. 9. 10.
11.
Bacha EA, Mathisen DJ, Grillo HC. Airway trauma. In: Westaby S, Odell JA, editors. Cardiothoracic trauma. London: Arnold; 1999. p. 265–79. Mathisen DJ, Grillo HC. Laryngotracheal trauma. Ann Thoracic Surg 1987;43:254–62. Couraud L, Velly JF, Martigne C, N’Diaye M. Posttraumatic disruption of the laryngotracheal junction. Eur J Cardiothorac Surg 1989;3:441–4. Schaeffer SD. The acute management of external laryngeal trauma. A 27-year experience. Arch Otolaryngol Head Neck Surg 1994;118:598–604. Burke JF. Early diagnosis of traumatic rupture of the bronchus. JAMA 1962;181:682–6. Symbas PN, Justicz AG, Ricketts RR. Rupture of the airways from blunt trauma: treatment of complex injuries. Ann Thorac Surg 1992;54:177–83. Kiser AC, O’Brien SM, Detterbeck FC. Blunt tracheobronchial injuries: treatment and outcomes. Ann Thorac Surg 2001;71:2059–65. Massard G, Rougé C, Dabbagh A, et al. Tracheobronchial lacerations after intubation and tracheostomy. Ann Thorac Surg 1996;61:1483–7. Mussi A, Ambrogi MC, Menconi G, et al. Surgical approaches to membranous tracheal wall lacerations. J Thorac Cardiovasc Surg 2000;120:115–8. Hulscher JBF, Hofstede E, Kloek J, et al. Injury to the major airways during subtotal esophagectomy: incidence, management and sequelae. J Thorac Cardiovasc Surg 2000;120:1093–6. Angood PB, Attia EL, Brown RA, Mulder DS. Extrinsic civilian trauma to the larynx and cervical trachea —
12. 13. 14. 15.
16.
17. 18. 19. 20. 21.
important predictors of long-term morbidity. J Trauma 1986;26:869–73. Chagnon FP, Mulder DS. Laryngotracheal trauma. Chest Surg Clin North Am 1996;6:733–48. Kelly JP, Webb WR, Moulder PV, et al. Management of airway trauma II: combined injuries of the trachea and esophagus. Ann Thorac Surg 1987;43:160–3. Wright CD, Wain JC, Donahue DM, et al. Tracheobronchial trauma: management of early and late airway disruption. [In preparation] Deslauriers J, Beaulieu M, Archambault G, et al. Diagnosis and long-term follow-up of major bronchial disruptions due to nonpenetrating trauma. Ann Thorac Surg 1982;33:32–9. Gaissert HA, Grillo HC, Lofgren R. Upper airway compromise after inhalation injury: complex strictures of larynx and trachea and their management. Ann Surg 1993;218:672–8. Moylan JA, Chan CK. Inhalation injury — an increasing problem. Ann Surg 1978;188:34–7. Adams C, Moisan T, Chandrasekhar AJ, Warpeha R. Endobronchial polyposis secondary to thermal inhalational injury. Chest 1979;75:643–5. Williams DO, Vanecko RM, Glassroth J. Endobronchial polyposis following smoke inhalation. Chest 1983;84:774–6. Perez-Guerra F, Walsh RE, Sagel SS. Bronchiolitis obliterans and tracheal stenosis. Late complications of inhalation burn. JAMA 1971;218:1568–70. Cros A-M. Lasers and the airway. In: Roberts JT, editor. Clinical management of the airway. Philadelphia: WB Saunders; 1994. p. 355–67.
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CHAPTER TEN
Tracheostomy: Uses, Varieties, Complications Hermes C. Grillo, MD
Tracheostomy Minitracheostomy Tracheostomy in Children Persistent Stoma
Tracheostomy Other than being a surgical technique, tracheostomy has many aspects that merit discussion; hence, I thought it best to consider these in a section of this book which is otherwise devoted to diseases of the trachea. Included elsewhere are historical notes on tracheostomy, surgical technique (see Chapter 22, “Tracheostomy, Minitracheostomy, and Closure of Persistent Stoma”), tracheostomy devices (see Chapter 38, “Tracheal Appliances” and Chapter 39, “Tracheal T Tubes”), and stents (see Chapter 40, “Tracheal and Bronchial Stenting”).
Indications For many years, tracheostomy was the primary means of providing emergency relief for upper airway obstruction. In the period following World War II, tracheostomy was considered to have three primary purposes: 1) emergency relief of airway obstruction, 2) management of secretions, especially after chest and central nervous system injury, and 3) to decrease respiratory dead space in order to improve ventilation. During the poliomyelitis epidemics of the early 1950s, tracheostomy was increasingly used to provide a route for administration of positive pressure ventilation for respiratory failure or impending respiratory insufficiency. This widespread use increased awareness of the many potential complications of tracheostomy and also introduced a new spectrum of lesions that resulted from intubation and mechanical ventilation. The immediate complications of tracheostomy, now very much reduced in incidence, are noted in this chapter, but the later complications, largely of postintubation origin, are dealt with in Chapter 11, “Postintubation Stenosis,” Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula,” and Chapter 13, “Tracheal Fistula to Brachiocephalic Artery.” Also considered in this chapter is the special topic of tracheostomy in children. Today, tracheostomy is rarely performed to provide an emergency airway. The airway is usually reestablished emergently by speedy insertion of an endotracheal tube, usually orally. Benign stenosis, such as that resulting from intubation, is best managed emergently by systematic dilation, without tracheostomy. Even in the presence of organic upper airway obstruction by a tumor, it is usually possible to slip an endotracheal
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tube into the airway, above or past an obstruction, in order to provide emergency ventilation until the obstruction can be dealt with bronchoscopically (see Chapter 19, “Urgent Treatment of Tracheal Obstruction”). For difficult intubations, a flexible bronchoscope is frequently of great help (Figure 10-1). The endotracheal tube is passed over the bronchoscope, which has been advanced beyond the obstruction. This technique is also valuable in patients with anatomically difficult airways or in those whose access is limited by severe cervical arthritis or temporomandibular arthritis, or by malformations. The rigid bronchoscope may also be a last resort for airway access, and in most cases, except the last mentioned, it can be introduced perorally by a skilled bronchoscopist. On rare occasions, an endotracheal tube may be threaded over a rigid pediatric bronchoscope, with a short length of proximal “pusher” tube also threaded proximal to the endotracheal tube (see Figure 10-1). With the aid of a straight bladed laryngoscope (Miller blade), the assembly is passed into the airway, the endotracheal tube pushed into the trachea, and the bronchoscope and “pusher” tube withdrawn. The laryngeal mask airway is a useful option if all else fails (see Figure 10-1). Roberts discusses the many aspects of these problems in Clinical Management of the Airway.1 Finally, if obstruction is high, a large bore needle or a small bore catheter may be inserted through the cricothyroid membrane to provide emergent oxygenation while either intubation or tracheostomy is accomplished. Many previously seen immediate complications of tracheostomy were incurred during the emergent placement of a tra-
A
B
C
10-1 Tools for the management of difficult intubations. From top to bottom. A, Endotracheal tube (ET) threaded over a flexible bronchoscope. B, Endotracheal tube over a pediatric rigid bronchoscope. Note the short segment of plastic “pusher” tube on the bronchoscope proximal to the ET. The assembly is introduced with a straight-bladed, open laryngoscope. The ET adapter is replaced after intubation is effected. C, Laryngeal mask airway, which is introduced to rest upon and seal over the larynx. FIGURE
Tracheostomy: Uses, Varieties, Complications
cheostomy tube under poor conditions. With tracheostomy now being almost always an elective procedure, these early complications, such as injury to carotid vessels or pneumothorax, have vanished. The second classic indication for tracheostomy, the management of secretions, has not been entirely replaced. However, with adequate humidification of the airway, effective employment of skilled pulmonary physiotherapy, endotracheal catheter suctioning, and more commonly, frequent flexible bronchoscopic aspiration, intubation is rarely necessary for secretions alone. Minitracheostomy, considered in detail later in this chapter, is an effective method for the management of persistent copious secretions. With the advent of positive pressure ventilatory support, tracheostomy is no longer used to facilitate ventilation by reducing dead space. A current major use of tracheostomy is as a route for mechanical ventilatory support. Endotracheal tubes are used for prolonged periods of time for ventilatory support. Although endotracheal tubes may produce their unique spectrum of complications, tracheostomy is often deferred for some time.2 No definitive studies are available to dictate a universally accepted management policy. If an endotracheal tube must be left in for more than 48 hours, it is often replaced with a more comfortable nasotracheal tube. Secretions are more easily managed by nursing staff through a tracheostomy tube than through an endotracheal tube. For this reason, as well as for patient comfort, and, importantly, in order to avoid serious injury to the glottis and subglottis, we usually replace an endotracheal tube with a tracheostomy tube in an adult in between 5 to 7 days, unless it appears that the patient may be weaned in a further short period of time. If it is clear that a patient will require prolonged mechanical ventilation, as in polyneuritis, then tracheostomy is done earlier. Tracheostomy continues to be useful to establish an airway temporarily or permanently wherever chronic obstruction presents, and definitive correction must be postponed or is not possible. In many such situations, a T tube will be preferable (see Chapter 39, “Tracheal T Tubes”).3 In the case of an obstructive benign stenosis which is accessible in the neck, it is mandatory that the tracheostomy or T tube stoma be located in the stenotic segment. In this way, further damage to the trachea is prevented, and if resection is later possible, the stoma and stenotic lesion will be simultaneously resected. Unfortunately, surgeons continue to place tracheostomies below such lesions, thereby damaging a further centimeter or two of the trachea (Figure 10-2A). Often, it becomes necessary to relocate such a misplaced stoma to the stenotic segment and allow the initial stoma to heal, in order to recapture this length of trachea and so facilitate or make possible later reconstruction. If a stenotic lesion is very low, even retrosternal, the stoma is carefully placed at the conventional level (second and third rings), leaving a generous segment of normal trachea between the stoma and lesion. The tracheostomy tube must then be long enough to pass through the dilated stenosis to splint it open. If the tracheostomy tube tip lies above the stenosis, it will only create an illusion of airway control (Figure10-2B). Additional indications for tracheostomy include complementary use with laryngeal anastomoses that may suffer transient glottic edema and for management of aspiration due to laryngeal dysfunction. Postoperative tracheal anastomotic complications may require tracheostomy (or T tube placement), sometimes for definitive management, but most often to permit resolution until reconstruction may again be safely considered (see Chapter 21, “Complications of Tracheal Reconstruction”). Tracheostomy should generally not be used for an obstructing tumor. Tumor is best managed in emergency by intubation past the tumor and then by the coring-out of tumor bronchoscopically (see Chapter 19, “Urgent Treatment of Tracheal Obstruction”). Definitive treatment then follows, either surgical resection or irradiation. Occasionally, under emergency circ*mstances in non-hospital settings, the need arises for external opening into the airway. Most often, the Heimlich maneuver accomplishes dislodgement of an acute supraglottic obstruction, such as that caused by an aspirated chunk of food. If this fails, and in the absence of laryngoscopic equipment, cricothyroidotomy should be employed. The cricothyroid membrane is the most superficial portion of the cervical airway. Even in obese individuals, this area can usually be palpated with the neck in hyperexten-
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sion. In my opinion, cricothyroidotomy should almost never be used as an elective route of airway intubation because of the likelihood of serious and possibly irreversible laryngeal damage. Emergency cricothyroidotomy is preferably converted to tracheostomy if its use will be prolonged. Jackson in 1921 cautioned against “high tracheostomy” (cricothyroidotomy) as a cause of subglottic stenosis (Figure 10-2C–E).4 The precept was challenged by Brantigan and Grow in 1976, although they later reported an incidence of subglottic stenosis following the elective procedure.5,6 Antecedent endotracheal intubation for a length of time appeared to predispose to such a stenosis.7,8 An overall incidence of at least 2% stenosis and up to 32% permanent voice changes followed cricothyroidotomy.7 Since there are few real advantages, if any, to cricothyroidotomy over tracheostomy, and since subglottic laryngeal stenosis is sometimes poorly correctable or impossible to correct, whereas nearly all postintubation tracheal stenoses are initially correctable, there is no reason to subject a patient to the hazard of cricothyroidotomy. The concern which led to the use of cricothyroidotomy, that is, separation of the stoma from median sternotomy for cardiac surgery, may be addressed by a few days of endotracheal tube ventilation to allow tissue planes to seal, thereby permitting tracheostomy to be done safely. However, the question of whether cricothyroidotomy poses a threat to median sternotomy remains controversial.9,10
Technique The technique of tracheostomy is described in Chapter 22, “Tracheostomy, Minitracheostomy, and Closure of Persistent Stoma.” Elective tracheostomy is best done in the operating room with adequate instruments and under ideal conditions. This minimizes the occurrence of both early and late complications. Percutaneous tracheotomes of various designs have appeared repeatedly over the decades, each seeming to lead eventually to a series of unnecessary and often serious complications, such as obstruction due to hemorrhage, damage to major blood vessels in the neck, intubation into the mediastinum rather than the airway, pneumothorax, tracheal wall laceration, and injury to recurrent laryngeal nerves. Recent techniques require placement of a guide wire or catheter and endoscopic confirmation of its location before plunging a tracheotome into the neck.11 A so-labelled “fingertip subcricoid minitracheostomy” (done at bedside) is a combination of a very small incision for dissection to the tracheal wall with wire-guided dilation and tube insertion, still generally called a percutaneous tracheostomy.12 With these precautions, it is hoped that many of the previously seen complications will not result. The presumed advantage over fully open procedures remains unsettled. Melloni and colleagues compared surgical versus percutaneous dilational tracheostomy prospectively in a consecutive series of 50 patients.13 They concluded that the percutaneous method was simpler and quicker, and had many fewer early complications, but had two late complications (malacia and stenosis), compared with no late complications surgically.
Complications The majority of immediate complications of tracheostomy incurred were often due to hurried performance of tracheostomy under inadequate emergency conditions, with poor definition of anatomic landmarks. These complications have largely disappeared.14 Hypoxia, which occurs during an urgent performance of tracheostomy and is sometimes accompanied by cardiac arrest, is eliminated when the procedure is done under elective conditions after establishment of an adequate airway. Laceration of the membranous tracheal wall can occur as a result of excessively forceful insertion of an inappropriate tube. The procedure of tracheostomy itself nowadays almost always follows establishment of an airway. A rare exception may be necessary, in separation of the cervical trachea following blunt trauma, where emergency bronchoscopy fails to reveal a channel to the distal trachea (see Chapter 9, “Tracheal and Bronchial Trauma”). In such a case, preparation is always made prior to endoscopy for emergent tracheostomy. Operative damage to structures such as the recurrent laryngeal nerves, the great vessels of the neck, and the esophagus has been
Tracheostomy: Uses, Varieties, Complications
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D
E
FIGURE 10-2 Errors in location of the tracheostomy. A, Placement of the stoma below a cervical stenosis lengthens the extent of damaged trachea. A needed stoma should be located in the stenosis, an already damaged segment. In some patients, it is judicious or necessary to relocate a stoma correctly and allow the prior stoma to heal, in order to recapture usable trachea for reconstruction. B, An insufficiently long tube that fails to pass through a low stenotic lesion fails in its purpose. The solution is not to lower the stoma but to use a longer tube. In urgent cases, a modified endotracheal tube may be used. C, D, The stoma should not be located, whether by intent or error, in the cricothyroid membrane or through the cricoid cartilage. E, A stoma located just below the cricoid, especially in a kyphotic patient, may erode the central cricoid cartilage and result in a partly intralaryngeal subglottic stenosis.
largely eliminated with adequate exposure, good lighting, adequate anesthesia, and precise surgical technique. Pneumothorax during tracheostomy has become very rare. It tended to occur in small children. Obese, short-necked, kyphotic individuals may still present technical challenges. Longer-term complications present chiefly as sepsis, hemorrhage, and obstruction. Additional complications include acquired tracheoesophageal fistula (TEF) and persistence of tracheal stoma. Sepsis of invasive or necrotizing type is surprisingly rare, even though all tracheostomies are soon contaminated, most
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often with Staphylococcus aureus (often a resistant strain) and Pseudomonas aeruginosa. Streptococcus and Escherichia coli are frequently present. This will occur despite sterile surgical technique and careful management of stoma, tubes, and suctioning. Antibiotics are not employed unless there is evidence of local invasion or pulmonary infection, to minimize overgrowth of other organisms. The contamination clears when the device is removed and the stoma is permitted to heal. Hemorrhage, obstruction at the laryngeal and tracheal level due to granuloma, stenosis, and malacia, TEF, and persistent stoma are detailed in subsequent chapters, and their surgical treatment described (see Chapter 11, “Postintubation Stenosis,” Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula,” and Chapter 13, “Tracheal Fistula to Brachiocephalic Artery”). Prevention is also stressed. Dysphagia and a tendency to aspiration, more pronounced in the elderly, may follow tracheostomy. Usually, this will ameliorate with time.
Minitracheostomy Matthews and Hopkinson described minitracheostomy as a method for removal of secretions from large airways while preserving glottic function for cough and phonation.15 A small-bore cannula (OD 5.4 mm) is placed into the trachea through the cricothyroid ligament for ease in suctioning. The technique is described in Chapter 22, “Tracheostomy, Minitracheostomy, and Closure of Persistent Stoma.” There is no interference with glottic competence, and the cough, which is stimulated by suctioning, is effective. The cannula may also be used for the intratracheal administration of medications and for access for jet ventilation. It may also be used in an emergency for temporary access to the airway. Minitracheostomy does not carry with it the potential complications of endotracheal intubation or of formal tracheostomy, since it does not interfere with the normal physiology of glottic function and cough, nor does it produce the same kind of potential for local tissue injury. It also seems far less likely to injure the subglottic larynx than conventional cricothyroidotomy does with placement of a full sized cannula. It is applicable to any patient who has excessive tracheobronchial secretions or ineffective cough. If a patient does not respond to the usual postoperative techniques for clearing of secretions, namely, chest physiotherapy, blind endotracheal suctioning, or flexible bronchoscopy after a limited number of trials, then minitracheostomy offers an additional avenue. It is best used early in patients in whom such difficulties may be anticipated. Minitracheostomy was effective when electively used prophylactically, to control sputum retention after lung operation in high-risk patients.16 A no. 10 French suction catheter is used for aspiration, and a small amount of saline (3–5 cc) is instilled prior to suctioning to stimulate cough and to loosen secretions. The cannula is otherwise capped to maintain humidification and to prevent drying of secretions. After initial placement, the catheter is suctioned frequently, then every 4 hours, and subsequently as needed. The cannula is well tolerated and does not limit the patient’s mobility, speaking, breathing, or swallowing. It does not stimulate aspiration, as sometimes a tracheostomy will. Suctioning through a minitracheostomy is more comfortable than blind endotracheal suctioning and it is certainly more comfortable than flexible bronchoscopy. Minitracheostomy has been particularly useful in postoperative patients who demonstrate difficulty in clearing secretions. In our experience, over 70% of the patients who required minitracheostomy responded to this technique alone.17 The rest of the patients required endotracheal intubation or formal tracheostomy because of progressive pulmonary insufficiency. Minitracheostomy does not provide an airway of sufficient diameter for spontaneous nonassisted ventilation with complete glottic obstruction. Although it may be used in an emergency situation for insufflation of oxygen, it can only be used as an airway with jet ventilation, using ventilators to provide the proper humidification, as described by Matthews and colleagues.18 Campbell and colleagues demonstrated no permanent changes in laryngeal function or adverse effects on the larynx following minitracheostomy.19
Tracheostomy: Uses, Varieties, Complications
Clinical Experience Wain and colleagues analyzed the first 56 patients in whom minitracheostomy was used at the Massachusetts General Hospital.17 Twenty-four of the patients were general thoracic, 9 were cardiovascular, 18 were general surgical, and 5 were medical. Their preceding surgical procedures and conditions included thoracotomy (20), esophagectomy (8), coronary artery bypass grafting (8), thoracic or abdominal aortic aneurysm repair (7), gastrointestinal (7), and subdural hematoma (1). Sixty minitracheostomies were placed, with repeat procedures performed at discrete intervals for different indications during complex hospital courses rather than because of malfunction of the minitracheostomy itself. The principal indication was excessive postoperative secretions in 46 patients. Twenty-five of these patients who were extubated immediately had minitracheostomy placed at a mean of 4 days after surgery, and in 21 patients who were ventilated postoperatively, the minitracheostomy was placed at a mean of 2 days after extubation. Additional indications were excessive postpneumonic secretions (5), preoperative secretions (4), and difficulty with standard blind endotracheal suctioning (4). In 1 patient, a minitracheostomy was used as an emergency means of establishing an airway until formal tracheostomy was done. The procedure was done at bedside in the intensive care unit in 32 patients and in a medical or surgical unit in 28 patients. Complications were seen in 1 in 6 patients. Intratracheal bleeding of significance occurred in 2 patients and emergency intubation was required. If intratracheal bleeding of significance occurs, an endotracheal tube must be placed immediately, over a flexible bronchoscope, before withdrawing the minitracheostomy catheter. Two cases of hemorrhage were probably related to injury to veins anterior to the cricothyroid membrane, with bleeding into the trachea, following removal of the cannula. If the cannula has been successfully placed in the trachea, it is left in place and the superficial hemorrhage is controlled by pressure or other conservative measures. Extratracheal placement of the catheter has been described by others, but it is our belief that careful attention to anatomic landmarks will prevent this. Serious complications may be recognized or prevented by performing flexible bronchoscopy routinely with every placement of a minitracheostomy tube.
Tracheostomy in Children The spectrum of diseases requiring intubation or tracheostomy in infants and small children differs from that of the adult. Congenital malformations of the nasopharynx, oral cavity, neck, larynx, and trachea or laryngeal hemangioma may be indications for urgent intubation. Acute laryngeal inflammatory processes including acute epiglottitis and laryngotracheal bronchitis, as well as acute laryngeal edema, sometimes due to allergic phenomena, may be emergency problems. Diphtheria is fortunately very rare and poliomyelitis has become so as well. The tiny size of the airway in the newborn or young child requires special skill in intubation. Nasotracheal tubes are preferred for comfort, especially if respiratory support is necessary. Cuffed tubes are not necessary for ventilation of infants and small children. In order to avoid erosive damage, the diameter of the tube is selected so that it will not impinge on the airway throughout its length. Severe and lengthy malacia or stenosis may result if a tube of excessive diameter remains in firm contact with the trachea for long. A tight fit at the glottic level may injure the cords and commissures, and result in stenosis. A tight fit at the cricoid level may produce subglottic stenosis.20 Infants and small children are carried for longer periods with endotracheal tubes, in order to avoid tracheostomy with its additional problems. On the other hand, the same trade-off of laryngeal injury is seen in children as in adults, although problems at the tracheal stoma may be eliminated (see Chapter 11, “Postintubation Stenosis”). Tracheostomy in an infant or child should be done over a previously established airway and under general anesthesia. Hendren and Kim recommended insertion of a rigid bronchoscope, which elevates the trachea and makes it easy to identify.21 A limited horizontal skin incision is preferred, placed below the
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cricoid. It is often necessary to divide the thyroid isthmus. The cartilaginous rings of the juvenile trachea are so tiny and soft that it seems preferable to divide vertically the third and fourth rings rather than the second and third as in the adult. It is important to avoid subglottic laryngeal injury. No tracheal wall is excised. Tracheostomy tubes fashioned specifically for infants and small children, such as those introduced by Aberdeen, are selected, avoiding tubes of too large a diameter (Figure 10-3).22 The curve of such tubes is unlikely to produce anterior granuloma or erosion from the tube tip. A degree of flexibility allows the tube to adapt to the curve of the airway. Modern humidification techniques permit the elimination of the inner cannula. The tube flanges slope upward so that the tapes do not tend to pull out the tube. The tubes are designed to accept a tracheostomy connector proximally, regardless of their basic internal diameter. With such precautions and with use of tubes especially designed for pediatric use, decannulation problems are reduced. The underlying lesion or a complication of the tracheostomy is probably the most common cause for difficulty in decannulation. Gradual progression to smaller tracheostomy tubes to smoothen the process of decannulation is sometimes advisable. The soft, thin wall of the infant trachea is easily deformed by a tracheostomy tube. In a number of children who have had tracheostomy tubes in place for some time, the anterior wall of the trachea just superior to the stoma becomes depressed by tube pressure. This deformity, together with thickening of the lower margin of the depressed flap, may cause obstruction on decannulation. Insertion of a small sized Montgomery Silastic T tube, with or without minimal excision of the tip of thickened scar at the lower end of the flap, restores the lumen. The tube is left in place for 3 months or more to allow the flap to become fixed in this more normal position. The T tube is then withdrawn and the child observed carefully for airway obstruction. A further period of splinting may be necessary. Residual cartilaginous structure must be
FIGURE 10-3
Pediatric tracheostomy tube. Pictured is a size 4 (ID 5.5 mm, OD 8 mm, length 4.6 cm) silicone tube (Shiley, Mallinckrodt Inc., St. Louis, MO). The curve of the tube and arrangement of the flanges are adapted for infants and small children, following the Great Ormond St. design. Note the standard size of the proximal adaptor end.
Tracheostomy: Uses, Varieties, Complications
present in the depressed flap to obtain permanent correction in this way. If the tracheal wall has been extensively replaced by scar, a stenosis will follow, requiring resection. It must be cautioned that T tubes are sometimes poorly tolerated in small children, probably because of the tiny diameters of their tracheae.3
Persistent Stoma Most tracheostomies close promptly after extubation. Indeed, reinsertion of a tracheostomy tube may become difficult within minutes or hours after its removal. In a few patients, the stoma persists, most often in those who have carried a tracheostomy tube for a very long time, who are aged or debilitated, who suffer from metabolic diseases, or who have received prolonged corticosteroid treatment. In most of these patients, the cutaneous epithelium has healed to the tracheal epithelium. A stoma is considered to be persistent if it remains patent 3 to 6 months after extubation. Some stomas contract after removal of the tube, but reach an end point without complete healing. The persistent stoma varies from full size to a sinus through which air and secretions escape. The persistent stoma may be a source of both difficulty and annoyance to the patient. Such patient may have to occlude the stoma in order to speak properly or to cough effectively, may be troubled by secretions, and breath with a whistling sound. I have not seen proven instances of increased susceptibility to pulmonary infection in such patients, but this concern is frequently raised. A patient with chronic obstructive pulmonary disease, dependent on nasal oxygen, found that she could breathe with much more comfort when the vent of the persistent stoma was closed. In most cases, closure of a persisting stoma is a minor procedure, easily done by techniques such as approximation of the strap muscle over the stoma. With a larger stoma, however, such a mesenchymal seal may lead to granuloma formation on the inner surface of the closed stoma. Our technique, described in Chapter 22, “Tracheostomy, Minitracheostomy, and Closure of Persistent Stoma,” provides immediate epithelial closure within the lumen and also corrects the cosmetic deficit that is usually present.23 Needless to say, closure is done only if the patient no longer requires a stoma for any purpose. If the patient still needs a stoma for suctioning, or if there is immediate likelihood of another tracheostomy, then closure is pointless. The single complication in over 30 patients was a subcutaneous hematoma in 1 patient with an undrained wound. This technique of closure has also been used in conjunction with resection of a more distal cuff stenosis of the trachea, where healing of skin to tracheal epithelium was present at the stoma. If granulation tissue rings the stoma, then this closure should not be attempted. In such patients, spontaneous closure will generally follow.
References 1. 2. 3.
4. 5. 6. 7.
Roberts JT. Clinical management of the airway. Philadelphia: WB Saunders; 1994. Stauffer JL, Olson DE, Petty TL. Complications and consequences of endotracheal intubation and tracheostomy. Am J Med 1981;70:65–75. Gaissert H, Grillo HC, Mathisen DJ, Wain JC. Temporary and permanent restoration of airway continuity with the tracheal T-tube. J Thorac Cardiovasc Surg 1994; 107:600–6. Jackson C. High tracheostomy and other errors: the chief causes of chronic laryngeal stenosis. Surg Gynecol Obstet 1921;32:392–8. Brantigan CO, Grow JB Sr. Cricothyroidotomy: elective use in respiratory problems requiring tracheostomy. J Thorac Cardiovasc Surg 1976;71:72–81. Brantigan CO, Grow JB Sr. Subglottic stenosis after cricothyroidotomy. Surgery 1982;91:217–21. Cole RR, Aguilar EA III. Cricothyroidostomy versus tracheotomy: an otolaryngologist’s perspective. Laryngoscope 1988;98:131–5.
8. 9.
10. 11. 12. 13.
Weymuller EA, Cummings CW. Cricothyroidotomy: the impact of antecedent endotracheal intubation. Ann Otol Rhinol Laryngol 1982;91:437–9. Stamenkovic SA, Morgan IS, Pontrefact DR, Campanella C. Is early tracheostomy safe in cardiac patients with median sternotomy incisions? Ann Thorac Surg 2000;69:1152–4. Curtis JJ, Clark NC, McKenney CA, et al. Tracheostomy: a risk factor for mediastinitis after cardiac operation. Ann Thorac Surg 2001;72:731–4. Hazard PB, Garrett HE Jr, Adams JW, et al. Bedside percutaneous tracheostomy: experience with 55 elective patients. Ann Thorac Surg 1988;46:63–7. Ciaglia P, Graniero K. Percutaneous dilatational tracheostomy — results and long-term follow-up. Chest 1992;101:464–7. Melloni G, Muttini S, Gallioli G, et al. Surgical tracheostomy versus percutaneous dilational tracheostomy. A prospective randomized study with long-term follow-up. J Cardiovasc Surg 2002;43:113–21.
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14.
15. 16.
17. 18.
Grillo HC, Mathisen DJ. Tracheostomy and its complications. In: Sabiston DC, editor. Textbook of surgery. 15th ed. Philadelphia: WB Saunders; 1997. p. 1815–20. Matthews HR, Hopkinson RB. Treatment of sputum retention by minitracheostomy. Br J Surg 1984;71: 147–50. Bonde P, Papachristos I, McCraith A, et al. Sputum retention after lung operation: prospective, randomized trial shows superiority of prophylactic minitracheostomy in high-risk patients. Ann Thorac Surg 2002;74:196–203. Wain, JC, Wilson DJ, Mathisen DJ. Clinical experience with minitracheostomy. Ann Thorac Surg 1990;49:881–6. Matthews HR, Fisher HBJ, Smith BE, Hopkinson RB.
19. 20. 21.
22. 23.
Minitracheostomy: a new delivery system for jet ventilation. J Thorac Cardiovasc Surg 1986;92:673–5. Campbell JB, Watson MG, Povey L, Shenoi PM. Minitracheostomy and laryngeal function. J Laryngol Otol 1988;102:49–52. Lindholm CE. Prolonged endotracheal intubation. Acta Anaesth Scand 1969;Suppl 33:1–131. Hendren WH, Kim SH. Pediatric thoracic surgery. In: Scarpelli EM, Auld PAM, Goldman HS, editors. Pulmonary disease of the fetus and newborn and child. Philadelphia: Lea & Febiger; 1978. p. 166–234. Aberdeen E. Tracheostomy and tracheostomy care in infants. Proc R Soc Med 1965;58:900–2. Lawson DW, Grillo HC. Closure of a persistent tracheal stoma. Surg Gynecol Obstet 1970;130:995–6.
CHAPTER ELEVEN
Postintubation Stenosis Hermes C. Grillo, MD
Characteristics and Origin of Lesions Prevention of Postintubation Lesions Clinical Presentation and Diagnosis Management and Results
Acute tracheal lacerations resulting from intubation with endotracheal tubes or tracheostomy tubes are described in Chapter 9, “Tracheal and Bronchial Trauma.” Late secondary lesions, principally stenosis of the larynx and trachea, are discussed here. Tracheoesophageal and tracheoarterial damage due to intubation are treated in Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula,” and Chapter 13, “Tracheal Fistula to Brachiocephalic Artery.”
Characteristics and Origin of Lesions Since the 1960s, the steadily increasing use of endotracheal, tracheostomy, and cricothyroidostomy tubes for the management of secretions, prevention of aspiration and, most importantly, delivery of mechanical ventilatory support for respiratory failure have produced a spectrum of upper airway lesions that range in location from the nostril to the lower trachea, and in severity from pharyngitis to complete obstruction of the airway or asphyxiating hemorrhage (Figure 11-1). Immediate and early complications of tracheostomy are described in Chapter 10, “Tracheostomy: Uses, Varieties, Complications.” The majority of these lesions may be avoided by elective performance, use of careful technique, and proper management of tracheostomy. The later complications detailed in this chapter at first seemed unavoidable and unpredictable; in large part, they no longer are. Despite great strides that have been taken in their prevention, postintubation lesions continue to be the most frequently seen surgical tracheal problems. Their clinical characteristics and nature must be made better known so that patients will not continue to suffer delay in recognition of the lesions and so that optimal treatment is given. The tracheal surgeon must become familiar with laryngeal lesions that result from intubation. Since most patients are initially ventilated through an endotracheal tube, the larynx may also sustain lasting laryngeal injury, even though the patient presents clinically with a tracheal lesion or a tracheostomy. Serious complications may result if a surgeon repairs the trachea without prior assurance of laryngeal competence. For example, an inadequate glottis that is not recognized because of the presence of a tracheostomy preoperatively, and is diagnosed only after tracheal reconstruction, may require either endotracheal intuba-
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11-1 The spectrum of postintubation laryngeal and tracheal lesions. Various combinations of these lesions are seen in a single patient. Focus on a laryngeal or tracheal lesion should not lead to overlooking a lesion elsewhere in the airway. TEF = tracheoesophageal fistula; TIF = tracheoinnominate artery fistula.
FIGURE
tion or another tracheostomy to provide a competent airway, pending glottic correction. Either mode of intubation may compromise the healing of a freshly repaired trachea. A brief catalogue of common postintubation laryngeal lesions follows.
Laryngeal Problems Following Intubation Laryngologic problems related to tracheal surgery are described in Chapter 35, “Laryngologic Problems Related to Tracheal Surgery.” In 1950, Briggs used a plastic endotracheal tube for 42 days to administer prolonged respirator therapy.1 It remains common practice to replace an endotracheal tube used for ventilator treatment in a much shorter period with a tracheostomy tube. A tracheostomy tube is more easily managed by nursing personnel, with less danger of obstruction and more comfort for the patient than an oro- or nasotracheal tube. Endotracheal intubation provides an airway promptly and can avoid tracheostomy when prolonged mechanical ventilation is unnecessary. It is because of this lessened temporal exposure that fewer cuff lesions were seen following endotracheal intubation than after tracheostomy. A cuff that exerts high pressure on the trachea, either because of its innate characteristics or its usage, is as damaging when on an endotracheal tube as when it is on a tracheostomy tube or a cricothyroidostomy tube. Lesions due to cuffs on endotracheal tubes are usually located higher in the trachea because the cuff is seated higher in the trachea than it is with a tracheostomy tube. Tracheal stomal lesions are obviously avoided if tracheostomy is not done. In using prolonged endotracheal intubation, however, the physician exchanges the absence of potential stomal complications for complications at higher levels (nostril, pharynx, and principally larynx). Although Briggs, in his original case, found only minor ulcerations over the arytenoid and in two small areas of the trachea at autopsy, a spectrum of more severe lesions occurs.2,3 In a series collected from the literature, Lindholm reported approximately 1 death in 120 children as a probable complication rate of prolonged endotracheal intubation.2 Most deaths occurred during the period of intubation as a result of obstruction of the airway and this probably reflects the small caliber of tubes
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necessarily used in small children. Laryngeal mucosal changes were prospectively observed, without exception, after prolonged endotracheal intubation. These were located on the medial sides of the arytenoid cartilages in the interarytenoid region and against the posterolateral portion of the cricoid. Changes in children were less pronounced. The lesions varied from superficial epithelial damage with only slight inflammation to deep ulceration. Donnelly detailed the histopathology of intubation.4 Within 48 hours, the perichondrium of the vocal processes and cricoid laminae were focally ulcerated, and the severity increased with time. Bergström and colleagues had earlier reported similar damage.5 Injury to the mucosa was seen by scanning electron microscopy, as early as 4 hours after endotracheal intubation.6 Lindholm found that 6 of 225 adult patients showed respiratory obstruction after extubation, and in 4 of these patients, the difficulty lay in the larynx.2 Five of 38 children had respiratory obstruction following extubation, and all required tracheostomy. The precise incidence of postintubation lesions of the larynx or trachea has never been satisfactorily determined, nor are such figures very meaningful, given the changing and varied standards of equipment and care both in time and place. The lesions continue to occur today. Nearly two-thirds of adult patients with erosive lesions healed by primary epithelization within a month. In a third of the patients, a granuloma formed during healing, located largely on the medial side of the arytenoid cartilages. In many cases, the granuloma regressed spontaneously in 1 to 10 months, with a median of 60 days. The symptoms of a granuloma are irritative cough, hoarseness, and transient sensations of suffocation. Granuloma also occurs on the anterior portion of the vocal cord. In his study, Lindholm found two children who formed fibrous scars with circumferential stenosis at the level of the cricoid and a third with a posterior commissural scar bridging the interarytenoid space. Localization of damage is likely related to the curve of the endotracheal tube. When a relatively straight or slightly arched endotracheal tube is reshaped by the patient’s airway, considerable force is exerted posteriorly against the medial sides of the arytenoid cartilages and the posterior surface of the cricoid (Figure 11-2).2,7 Another factor may be movement between the larynx and endotracheal tube. More prolonged exposure to pressure by the tube seems to lead to a greater incidence and depth of injury. Lindholm recommended a preshaped tube with a gently curved right angle to try to avoid such pressure. Subglottic erosions at the cricoid level appear to be caused in large part by tubes that are too large for the particular airway. This is supported by the observation of a probably greater frequency of stenosis at this level in women and smaller males, who tend to have smaller airways. Since the cricoid cartilage is nor-
11-2 Pressure on the larynx is exerted by an endotracheal tube, chiefly posteriorly, against medial arytenoid cartilages and posterolateral cricoid. Adapted from Lindholm C-E.2
FIGURE
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mally the only complete cartilaginous ring in the upper airway, the absolute limit of the airway’s diameter is set by that cartilage. If pressure results only in edema, the condition is reversible. If ulceration occurs following pressure, then a cicatricial stenosis may result during healing. Fortunately, the cricoid cartilage is a rather rugged structure, and it is unusual for it to be eroded very deeply or through its full thickness by an endotracheal tube. This probably accounts for the fact that prolonged stenting of cicatricial lesions in this region may occasionally lead to success, in contrast to the usual failure of stenting for circumferential lesions in the trachea. It may also account for the greater potential for occasional success in laser treatment of obstructive lesions at this level, in contrast with the trachea where laser treatment often fails. Vocal cord disturbances are common in patients who have undergone prolonged endotracheal intubation. In a small percent, these disturbances may go on for over a month, but frequently regress. Permanent laryngeal paresis does occur in a small number of patients, presumably due to inflammatory involvement of recurrent laryngeal nerves. Such disability is likely to be unilateral. Postmortem studies have shown the precise location of postintubation ulceration or necrosis of the larynx in 33 patients to be as follows: 3 had ulcers in the interarytenoid area, 26 had ulceration or necrosis on the medial side of the arytenoid cartilages, and 33 ulcerative lesions were seen on the inner posterolateral part of the cricoid.2,5 Eight of 26 patients also had tracheal lesions. In other reported series, the incidence of subglottic stenosis of the larynx following prolonged endotracheal intubation in children has run from 0 to 8%, with one series reporting as high as 20%. In clinical practice, one may see the following at the glottic level: arytenoid fixation, interarytenoid posterior commissural scar preventing separation of the cartilages, anterior commissural stenosis, and vocal cord thickening, ulceration, or granulomas. Unilateral vocal cord paralysis that is present as a consequence of endotracheal intubation, tracheostomy, or prior surgical procedures may affect the adequacy of the glottic aperture or contribute to aspiration on deglutition. The rare presence of a bilateral cord paralysis, most often post-traumatic or postsurgical, may cause airway inadequacy or aspiration. Subglottic intralaryngeal narrowing due to cicatricial stenosis may begin immediately below the glottis or at any level below that. It is often maximal at the cricoid level for the reasons explained. Often, this subglottic laryngeal stenosis is confluent with a stenosis that extends into the upper trachea. In 50 patients treated surgically for postintubation subglottic stenosis with such a major component in the larynx, 31 resulted from endotracheal intubation alone, 16 from high or eroded tracheostomy, and 3 from cricothyroidostomy (Figure 11-3).8 The obvious statement must be made that wherever a foreign body impinges forcefully on the airway, whether a stoma is present or not, erosion may occur and be followed by cicatricial stenosis. Cuff lesions are common to all types of tubes and independent of them, except with regard to the level of cuff impingement. Stenotic injuries unique to endotracheal and cricothyroidostomy tubes are necessarily within the larynx. The stomal lesion from a tracheostomy lies within the trachea, except when a stoma is placed too high or, more often, in an older kyphotic patient, where erosion of the anterior cricoid occurs. “Subglottic stenosis,” that is, laryngotracheal stenosis beginning below the cords and usually extending into the upper trachea, is circumferential following injury from endotracheal tubes (see Figure 11-3A and Figure 28 [Color Plate 15]). Those stenoses that are caused by cricothyroidostomy tubes or eroding tracheostomy tubes may be largely anterior or also circumferential (see Figures 11-3B,C). In all cases, we are dealing with laryngotracheal “bed sores,” since pressure necrosis is the primary damaging factor (Figures 11-4A–E). Brantigan and Grow proposed eliminating tracheal stomal stenosis by transferring the stoma to the cricothyroid membrane.9 Although they were fortunate that their series had no stomal complications (or cuff stenosis), clearly, this approach merely transfers the location from the trachea to the larynx (Figures 11-5A,B).3,8 Subsequent studies by these and other authors showed that cricothyroidostomy contributes to a significant and often irreversible subglottic stenosis, with an overall airway coimplication incidence of up to 52%.10 Kuriloff and colleagues pointed out that the short length of the cricothyroid membrane (5 to 12 mm)
Postintubation Stenosis
11-3 Origin of a subglottic postintubation laryngotracheal stenosis. A, Circumferential cricoid erosion by the endotracheal tube. B, Proximal erosion of cricoid by a high tracheostomy in a kyphotic individual. Anterolateral stenosis results. C, Subglottic stenosis secondary to anterior cricoid erosion by the cricothyroidostomy tube residing in the cricothyroid membrane.
FIGURE
A
B
C
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A
C
B
D
11-4 Subglottic postintubation stenoses. A, Roentgenogram of the larynx and upper trachea. A slight 25-year-old man was ventilated with an endotracheal tube for head injury. Upper arrow indicates the vocal cord level. Lower arrow is at the cricoid level, also the point of maximum stenosis. The entire subglottic space is markedly narrowed. B, Laryngoscopic view just below the glottis in the same patient. The stenosis is severe, irregular, but circumferential. The glint of a tracheostomy tube is seen distally. Tracheostomy was necessary prior to laryngotracheal resection and reconstruction. C, Laryngotracheal stenosis following ventilation via “high tracheostomy,” probably cricothyroidostomy, after repair of ruptured chordae tendineae in a 71-year-old man. Lateral cervical roentgenogram. Arrows indicate severe constriction of the lower larynx and trachea. D, Airway restoration following laryngotracheal reconstruction. Normal lumen is restored (arrow). See also Figure 28 (Color Plate 15). FIGURE
Postintubation Stenosis
E
FIGURE 11-4 (CONTINUED)
E, Surgical specimens from a young woman who was ventilated after multiple injuries for a long period via an endotracheal (ET) tube, continued via a tracheostomy tube. Subglottic stenosis from ET tube injury is shown at left and result of the combined ET cuff and tracheal stomal injury is on the right.
is less than the outer diameter of most commonly used tracheostomy tubes (10 to 12 mm).10 Although most purely tracheal stomal stenoses can be corrected surgically on a first attempt with almost certain success, many laryngeal lesions are not correctable or only partially correctable from the outset. I, therefore, deplore use of elective cricothyroidostomy for ventilation. It should also be noted that concern about contaminating sternotomy incisions after cardiac surgery, by an immediately adjacent tracheostomy tube, may be obviated by using an endotracheal tube for ventilation for a few days. If tracheostomy is needed subsequently, tissue planes will have already sealed. Preexisting neurologic deficits unrelated to the postintubation injury, particularly in patients who have suffered central nervous system trauma, may affect protective laryngeal reflexes and result in aspiration on deglutition. Such a deficit may preclude airway repair since tracheostomy may then be necessary to protect the airway and to clear secretions. Such functional deficits must be identified prior to a tracheal reconstruction.
Obstructive Lesions of the Trachea Obstructive lesions of the trachea following intubation occur at four levels, depending on the source of injury. At each level, one or more distinct lesions may produce obstruction. The levels are 1) stomal, 2) the site where the inflatable cuff rested, 3) the segment between the stoma and the level of the cuff, and 4) the locus where a tip of the tube may impinge on the tracheal wall (see Figure 11-1). Lesions at Stomal Level. Since tracheostomy creates a defect in the wall of the trachea, whether the opening is made by a vertical, horizontal, cruciate, or T incision, by excision of a segment or segments of carti-
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lage, or by turning a flap, some scarring is inevitable during healing. Long after healing is complete, inspection, both bronchoscopically and radiologically, will demonstrate dimpling or deformity, an anterior shelflike projection, or softness of the anterior wall at the site of prior tracheostomy. A surprising degree of asymptomatic narrowing may occur. Nearly 50% narrowing of the cross-sectional area of the trachea, or even more, is necessary before a sedentary person experiences dyspnea. Three stomal lesions, seen alone or in combination, may cause obstruction. These are 1) granuloma, 2) a posteriorly depressed flap of tracheal wall above the stoma, and 3) anterolateral stenosis. Granulation tissue forms at the stoma before and during healing. Granulomas may be noted weeks or months after extubation. As healing progresses, ebullient granulation tissue may form on the inner surface of the trachea at the site of the stoma and become sufficiently bulky to obstruct the airway. Accumulation of this type of papillomatous granulation tissue often occurs in conjunction with deformity at the healing stomal site (see Figure 11-5). If a large granuloma is already present, immediate airway obstruction may follow removal of the tracheostomy tube. The curve of the tracheostomy tube may produce a depressed tracheal wall flap just above the stoma. The tip of the flap may be thickened or granulomatous, but in most cases, this alone is insufficient to cause serious obstruction when the tube is withdrawn. When the tracheostomy tube has been in place for a long time, the upper flap may remain positioned posteriorly and produce partial or even subtotal obstruction (see Figure 11-1). This tissue may even become calcified prior to removal of a long-standing tracheostomy tube. We do not know whether this flap effect can be avoided by choice of incision for tracheostomy. It seems to occur most commonly in children who have had prolonged tracheostomy, perhaps because of the thinness and pliability of the juvenile trachea.
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11-5 A, Anteroposterior tomogram of the larynx showing stenosis and granuloma (large arrow) at the site of cricothyroidostomy. Note the proximity of the vocal cords just above the lesion. B, Lateral view showing narrowing of the airway, deformity, and large granuloma. The “O” marks the stomal site on the skin. Laryngotracheal resection and reconstruction was necessary. FIGURE
Postintubation Stenosis
The most common lesion of significance at the stomal level is anterolateral stenosis. Following removal of the tracheostomy tube, the patient gradually develops obstructive symptoms. The patient is found bronchoscopically to have an A-shaped stricture with an apex anteriorly, which involves the anterolateral walls of the trachea (Figures 11-6A,B and Figures 24 and 25 [Color Plate 14]). The membranous wall is usually spared but irritative granulation tissue may be present posteriorly in some cases (Figure 11-6C). The membranous wall may be shortened by deformity of the lateral walls, which are pulled together by the anterior scar. Stomal stenosis results from cicatricial healing of what was or has become a large stomal defect (Figure 11-7). A number of factors appear to play an etiologic role. Occasionally, a surgeon makes a much too generous opening in the tracheal wall, failing to realize that loss of tracheal substance will ultimately be healed by the natural process of contraction of the scar. Tracheostomies probably erode toward the size of their inlying tracheostomy tube due to local pressure necrosis, no matter how the stoma is made. Any opening which is larger than this may only add to the destructive process. All tracheal stomas are inevitably contaminated bacterially. Although invasive sepsis is not frequent, bacterial activity may lead to further local tissue destruction. The most important factor is the weight of unsupported tubing which levers the tracheostomy tube against the margins of the stoma, producing pressure necrosis. Evidence to support this was the decrease in incidence of stomal stenosis in a single respiratory care unit, before and after the introduction of lightweight swivel connectors to tracheostomy tubes.11 Careful suspension of tubes and connectors has essentially eliminated such lesions at Massachusetts General Hospital (MGH). As noted earlier, an especially complex stomal lesion results if the cricoid cartilage is eroded by upward pressure of the tracheostomy tube. If the anterior cricoid cartilage loses its integrity, anterior subglottic laryngeal stenosis occurs in conjunction with upper tracheal stenosis. Even if the first tracheal ring has not been mistakenly divided during tracheostomy, a tube that impinges against it may erode through it and into the cricoid cartilage. This is most likely to occur in older patients with a degree of kyphosis, where hyperextension of the cervical spine fails to draw the larynx far above the sternal notch. Although the tracheostomy tube may be correctly placed at the level of the second tracheal ring, it may have to arch across the cervical tissues to reach the skin surface, exerting pressure against the cricoid (see Figure 11-3B). It is important to recognize the extent of such lesions prior to surgery, since the technique of repair of a purely tracheal stenosis is very different from that for subglottic laryngotracheal stenosis (see Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection,” and Chapter 25, “Larygotracheal Reconstruction”). A comment on terminology is in order. “Subglottic” is often used to describe lesions anywhere from just below the vocal cords to the upper or even midtrachea. Surgical problems and prognosis are quite different at different levels. I prefer to describe a lesion that lies in the larynx between the vocal cords and the lower border of the cricoid cartilage as a subglottic lesion (intralaryngeal). If the upper border of the lesion lies just below the lower border of the cricoid cartilage, it is clearly an upper tracheal lesion, and laryngotracheal spans the subglottic level and upper trachea. Infrastomal Obstructive Lesions. The principal infrastomal lesion that results from intubation for respiratory support is tracheal stenosis at cuff level or cuff stenosis. It is the most common lesion complicating modern respiratory care and it is clearest in the minds of most physicians (Figure 11-8 and Figures 26 and 27 [Color Plates 14 and 15]). The origin of stenosis at cuff level was obscure when the lesion was first recognized. It originates from circumferential erosion of the tracheal wall due to the pressure of the cuff and is common to all forms of access to the trachea; endotracheal tubes, tracheostomy tubes, or cricothyroidostomy tubes (Figure 11-9). In the extreme, a tracheo-innominate artery fistula can result if erosive pressure is maximum anteriorly, or a tracheoesophageal fistula if the erosion penetrates posteriorly. Conventional high-pressure cuffs formerly in use, whether on endotracheal tubes or tracheostomy tubes, exerted enormous pressures on the tracheal wall. They almost uniformly produced some degree of tracheal injury
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within 48 hours of placement. The depth and severity of damage is roughly, but not uniformly, related to the duration of exposure to pressure injury. The key etiological factor in the production of stenosis is pressure necrosis caused by the cuff, compressing the tracheal mucosa and, later, the deeper structures of the tracheal wall. The principal evidence supporting these conclusions has been derived from autopsy study of patients who received ventilatory support,12,13 from prospective studies by direct visualization of the tracheae of patients receiving ventilatory assistance,11,14 and from experimental production of identical
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B FIGURE 11-6 A, Surgical specimen of stomal stenosis showing the typical triangular lumen with apex anterior. A granuloma is also present where the stoma lay. The membranous wall is smooth. B, Longitudinal view of the same specimen demonstrating that the tracheal walls are pulled together by the cicatricial closure of the stomal defect. C, Another specimen showing stomal stenosis with active inflammatory granulation tissue everywhere, including the membranous wall of the trachea. An endotracheal tube had been wedged into the stenosis on several occasions. Also, see Figures 24 and 25 (Color Plate 14).
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11-7 Evolution of stomal stenosis. Cross-sectional diagrams of the trachea at the stomal site. A, The tracheostomy tube may be leveraged against the stomal margins, causing their erosion. B, The resulting large stoma exhibits granulations and inflammation at its margins. C, Contraction of scar tissue forming across the defect pulls the tracheal stomal margins to the midline. D, The result is an A-shaped lumen. The posterior wall is also often shortened. Scar tissue is present, in most anteriorly only. FIGURE
lesions.15 Correlation of these observations with surgically removed specimens further supports this thesis.16 Final confirmation is that removal of excessive cuff pressure has eliminated the lesions in a very large number of patients at risk over a long period of time, at MGH. Cooper and Grillo examined the tracheae of 30 patients who died while receiving ventilatory assistance through cuffed tracheostomy tubes as well as 4 additional patients who had received such assistance through cuffed endotracheal tubes only for short periods of time.12 Both metal and plastic tracheostomy tubes and plastic and rubber cuffs had been used. At that time, all cuffs were of designs that produced high intracuff pressures. A consistent pattern of tracheal damage was observed, with the major damage located at the site of the cuff. The period of mechanical ventilation and the degrees of damage generally correlated. Superficial tracheitis and fibrin deposits appeared within 48 hours of placement of the tube. Small, shallow ulcerations were then seen, overlying the cartilaginous rings. With time, the size of the ulcers increased and cartilages were exposed. The inflammatory process spread laterally and deeply, followed by fragmentation of cartilage (Figure 11-10). The tracheal wall in many cases bulged where the balloon was located. These lesions usually began approximately 1.5 cm below the inferior margin of the tracheostomy stoma and extended downward for a length of about 2.5 cm, that is, the location of the cuff. Usually, between two to four cartilages were completely bared in time. Eventually, segments of cartilage sloughed and, in advanced cases, the balloon site was completely devoid of cartilages. Severe damage was observed between 10 days to 2 weeks after placement of the cuff. Additional ulcerations were occasionally seen, corresponding to the tip of a tracheostomy tube below the cuff injury. Changes that occurred at cuff sites from endotracheal tubes compared closely to those seen from cuffs on tracheostomy tubes with similar duration of intubation. These lesions were located more proximally in the trachea, since the cuff of an endotracheal tube is sited more proximally than that from a tracheostomy tube (see Figure 11-9). Microscopic examination elaborated the progressive changes that were found grossly (Figure 11-11). Acute inflammation and fibrin appeared early. Microscopic ulceration followed, overlying the cartilaginous rings where the mucosa had been compressed between the balloon and the underlying cartilage (see Figure 11-11A). As the ulcers deepened, the surfaces of the cartilages were bared, and inflammatory degeneration ensued. Inflammatory cells infiltrated beneath the cartilages (see Figure 11-11B). Fragmentation of
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B FIGURE 11-8 A, Cuff level stenosis. The lesion is circumferential. The lumen is less than 5 mm in diameter, the level at which the patient became dyspneic on bed rest while recovering from multiple orthopedic injuries and after a remote period of ventilation. B, The same lesion just prior to resection (arrows). A Penrose drain lies beneath the stenotic segment. C, Photomicrograph of the lesion. Dense fibrosis and partial cartilaginous destruction are evident (hematoxylin and eosin; ×25 original magnification).
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cartilage occurred next (see Figure 11-11C) and, finally, only an ulcerated bed remained where the cartilage had sloughed (see Figure 11-11D). In some cases, the tracheal wall was totally replaced with granulation tissue as repair competed with erosion (see Figure 11-11E). At the margins where the epithelium remained, squamous metaplasia was evident. Although the membranous wall sometimes escaped changes as severe as those in the noncompliant cartilaginous portion of the tracheal wall, severe erosive inflammatory changes were produced nonetheless. The circumferential lesion reflected circumferential pressure injury. In several cases, the membranous wall was reduced to paper thinness, and there were inflammatory changes in the adjacent esophageal wall. Complete fistulization occurred in other specimens. Florange and colleagues reported similar pathologic findings.13
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FIGURE 11-9 Evolution of a cuff stenosis. Excessively high pressure exerted on the trachea produces circumferential erosion, which heals by cicatrization of granulation tissue. The resulting scar is circumferential. A, Cuff lesion from an endotracheal tube. B, Cuff lesion from tracheostomy tubes. The lesion is usually lower than that from an endotracheal tube.
These changes were compared with a group of surgically resected specimens of cuff stenosis, where a circumferential ring of dense fibrous tissue resulted at 1 to 3.5 cm below the tracheostomy site. In these resected specimens, the residual effective airway often measured 2 to 5 mm in diameter. Externally, the area of stenosis was usually demonstrable, frequently hourglass in shape, but without external indication of the extreme narrowness of the minimal internal airway (see Figures 11-8A,B). In longstanding cases, metaplastic squamous epithelium was present. In most, the scar tissue of the stenotic ring remained unepithelialized. In some, residual pieces of tracheal cartilage were present in a greater or lesser degree (Figure 11-12). In fully advanced cases, not even vestigial remnants of tracheal architecture were identifiable. Patients with cuff stenoses following endotracheal intubation often had marked cicatricial stenosis, but relatively intact outer cartilages, presumably because the length of exposure to pressure had been too brief to necrose the cartilages completely. A striking finding was that all patients with the then-conventional cuffs in place had notable changes. These findings were confirmed in a prospective study by Andrews and Pearson, who examined the tracheal wall endoscopically through the stoma at the time of removal of the tracheostomy tube.11 We made similar endoscopic observations in a study of the comparative effects of standard cuffs and an experimental low-pressure cuff.14 Many etiologic possibilities for these lesions had earlier been suggested, including the influence of sepsis, the fact that many patients had periods of hypotension during their illness which could impair circulation in the compressed mucosa, damage by toxic materials in the tubes, and cuffs and from gas sterilization.17 Shelly and colleagues questioned the effect of systemic hypotension, on the basis of animal experiments.18 Experimental reproduction of the lesions helped to clarify these questions.15 Murphy and colleagues attempted to reproduce the lesions in dogs, but were able to do so only with the combination of cuff and tracheostomy.19 We placed short segments of Portex endotracheal tubes with cuffs perorally into the tracheae of dogs, and fixed them with percutaneous wires.15 Tracheostomy was avoided to minimize infection. The tubes were clean but not gas sterilized. The cuff was inflated just sufficiently to provide a seal at 25 cm of water ventilatory pressure, and this pressure was then maintained throughout the experiment. Destructive lesions were uniformly produced within 1 week (Figures 11-13A,B). Removal of the tube was
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FIGURE 11-10 Postmortem tracheal specimens of patients ventilated with high-pressure cuffs then in use. A, Trachea opened posteriorly. Portex tube and cuff are evident. Ventilation was for 19 days. The patient was age 55 years. B, Ulceration and loss of cartilage at the cuff site. C, Trachea of a 69-year-old woman, ventilated for 16 days. Metal tracheostomy tube with latex cuff are evident. D, Mucosa is destroyed at the cuff site and cartilages are bared and fragmented.
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11-11 Photomicrographs from postmortem specimens of tracheae injured by ventilation using high-pressure cuffs. Tracheal lumen is above in A, B, and C. The lumen is at the right in D and E. A, Erosion of mucosa over cartilages due to cuff compression. Submucosal inflammation. B, Cartilages now bared by progress of mucosal and submucosal necrosis. Lamina propria has been thickened by inflammation. C, Necrosis of cartilage follows with increased inflammatory intensity in surrounding tissues. D, Total destruction of cartilages occurs. Inflammation extends deeply into tracheal wall. E, The tracheal wall is now essentially replaced with inflammatory tissue and beginning reparative response of granulation tissue (hematoxylin and eosin; ×25 original magnification). FIGURE
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FIGURE 11-12
Photomicrograph of transverse section of a fully developed cuff stenosis. A fragment of degenerated cartilage is essentially the only immediately recognizable remnant of the tracheal architecture. Otherwise, a ring of scar tissue has replaced the trachea, and its contraction leaves only a very reduced lumen. Mucosa is lacking (hematoxylin and eosin; ×11 original magnification).
followed by cicatricial stenosis and airway obstruction (Figure 11-13C). Although the experimental and clinical evidence do not rule out possible additive contributions by other factors mentioned as well as others unknown, the principal common denominator appears to be necrosing pressure on tissue. We saw no cuff stenoses in over 5 years and in many hundreds of patients, following the design and introduction of a latex low-pressure cuff, although no other factors changed during this period. Since then, despite the necessary but careful use of plastic low-pressure cuffs, since latex cuffs are not available, no cuff stenoses have been produced at MGH in thousands of ventilated patients. Varying degrees of tracheitis occur in the segment between the level of the stoma and the level of the cuff. The segment is usually short, but it lies in close proximity to two areas of damaging influences. In many cases, secretions puddle above the cuff, despite intermittent deflation. Heavy bacterial colonization is routine around tracheostomies. Varying degrees of gross and microscopic inflammation are seen in this segment of trachea. The cartilages may be thinned and inflamed while the mucosa, although inflamed, is intact. At operation, the tracheal wall at this point may be markedly inflamed and its architecture partly destroyed. This becomes evident once part of the trachea is detached from surrounding supporting tissues. Tracheal malacia occurs in this segment and is demonstrable fluoroscopically or bronchoscopically. Such changes can be of great importance in planning surgical excision of cuff stenoses, since the segment of trachea requiring removal may be almost double the length apparent in preoperative static images of a cuff stenosis. In a few patients, the area of cuff damage itself may be primarily malacic rather than firmly stenotic, producing valve-like obstruction on deep breathing or coughing (Figures 11-14A,B). Routine tracheal x-rays may show only slight or no deformity at cuff level. Functional obstruction becomes evident only when deliberately sought for fluoroscopically or during an awake flexible bronchoscopy. In these patients, cartilaginous rings are absent, and the fibrous wall is covered with squamous metaplastic epithelium (Figure 11-14C). The evolution of malacia rather than fibrous stenosis at cuff level has not been explained.
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Lesions may overlap and it may be difficult to ascertain the precise etiology, especially if a stenosis appears late. In obese or aged individuals, for example, the cuff may reside immediately within the stoma, with confluence of stomal and cuff injuries. Multiple tracheostomies also serve to confuse the issue, since records of prior treatment are often imprecise. Stenosis resulting from prior endotracheal intubation may be lost sight of after a series of therapeutic tracheostomies and laser treatments. Occasionally, trachiectasis occurs. In a few patients with persistent respiratory failure from chronic lung disease, who are managed for lengthy periods of time with conventional equipment, the requirement for volume of air to seal the cuff gradually increases. Generally, high ventilatory pressures are needed. The trachea in one patient required a balloon volume of approximately 200 cc for a seal. Granuloma formation at a site of ulceration by the tip of a tracheostomy tube can also cause obstruction, although rarely. With the older type of high-pressure cuffs that expanded eccentrically, the tip of the tracheostomy tube could easily be angulated against the tracheal wall. A tracheostomy tube with a 90˚ angle accentuates this possibility. This also happened in children where no cuff was used at all, where slight angulation of a tracheostomy tube levered the tip against the tracheal wall (Figure 11-15). After the tube is removed, an ulcer may heal, with profuse connective tissue formation, producing an inflammatory granuloma. The incidence of such lesions in children has diminished remarkably with the development and use of improved pediatric tracheostomy tubes (see Chapter 10, “Tracheostomy: Uses, Varieties, Complications”). Granulation tissue may form around the lower end of a tracheostomy tube while a patient is still on a ventilator. This occurs most often in patients who have long been on mechanical assistance, who have
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11-13 Experimental production of cuff stenosis in dogs. A, Segment of the endotracheal tube with high-pressure (standard) cuff after 7 days of exposure to the seal at standard ventilatory pressure. B, Mucosal destruction has occurred, cartilages are bare, and the trachea is distended. C, In another specimen, 13 days of exposure has produced copious granulation tissue, replacing the normal tracheal structure. FIGURE
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C 11-14 Diagnostic images from a 38-year-old woman with attacks of dyspnea and changes of voice worsening over a year, following emergency tracheostomy and ventilation for obstructive laryngitis. A, “Spot film” from fluoroscopy demonstrating an apparently normal upper tracheal diameter (arrows). The adducted vocal cords and subglottic larynx are clearly defined above. Symptoms were thought to be of psychiatric origin because of similar static x-rays of the trachea elsewhere. B, Fluoroscopic picture of the segmental malacic tracheal collapse (arrows) on cough. Resection of the damaged trachea fully relieved her complaints. C, Surgical specimen from a 70-year-old woman who was twice ventilated for long periods for respiratory failure. Circumferential cuff lesion was principally malacic, with some fibrosis. FIGURE
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diffuse tracheitis, and who may have significant injury at cuff level. Bronchoscopic removal of granulations provides only transient relief. A longer tube or one with a different angle may be inserted, but the danger remains that the process will extend distally, ultimately to the carina, unless the patient can be weaned. Granulations in a supracarinal location present a special hazard, since a tube may slip back only a few millimeters and obstruction from the granulations may slowly form below the tip of the tube while the physician has an illusion of safety.
Incidence It has been difficult at any time to establish the incidence of lesions that follow tracheal intubation. In 1967, 17% of a vulnerable population from a respiratory care unit at the Massachusetts General Hospital developed clinical evidence of upper airway obstruction. This selected population consisted of survivors of relatively prolonged treatments of the most severe respiratory failures and the study occurred in the era preceding the development of low-pressure cuffs. Large numbers of patients who had received respiratory support for lesser problems, often through an endotracheal tube, were not included. The figure compared quite closely with the range then described from other institutions: 20% from the Toronto General Hospital with a similar population, 12% of a group of cardiac surgical patients from Mount Sinai Hospital in New York, and 16% of a group of 50 patients from Australia. Harley attempted to establish the incidence of laryngotracheal stenosis following tracheostomy and assisted ventilation, by analyzing reported series.20 The range was from 0 to 22%, with an average of 3.27% for 3,793 tracheostomies. Introduction of low-pressure cuffs of varying efficacy and closer attention to avoidance of stomal erosion greatly diminished the occurrence of injury in succeeding years. Following introduction of our prototype of the low-pressure latex cuff, cuff stenosis vanished at Massachusetts General Hospital. Careful attention to the use of currently available plastic large-volume, low-pressure cuffs has continued this record. Stomal strictures were reduced to well under 1% (see “Prevention of Postintubation Lesions” below). In recent years, further attention to tube support has eliminated these lesions as well. Unfortu-
11-15 Obstructing granuloma in a child, caused by erosion by the tip of a tracheostomy tube. The angle of many adult tubes is inappropriate for pediatric use. Bronchoscopic removal of the granuloma sufficed. FIGURE
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nately, this is not universally true, for reasons described later. Postintubation lesions of all types continue to be produced worldwide, and they continue to be the leading indication for tracheal surgery. Tracheoesophageal fistula (TEF) and tracheo-innominate arterial fistula resulting from intubation and ventilation are described in Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula,” and Chapter 13, “Tracheal Fistula to Brachiocephalic Artery.”
Prevention of Postintubation Lesions Prevention of all postintubation lesions of the trachea is unlikely until totally new methods of respiratory support are developed that do not require a foreign body in the trachea as part of the support system. Noninvasive techniques remain in the future.21 Enough has been learned about the origin of postintubation lesions so that their frequency has been markedly reduced and further improvement is to be expected.
Stenosis at Stomal Site Stomal stenosis may be minimized or avoided altogether by attention to details, performance, and management. First, the surgeon should make no larger an opening for the tracheostomy tube than is necessary. The tube should not be too large for the particular patient. Its curve should be appropriate. In order to minimize the size of the stoma and destruction of tracheal tissue, I prefer a simple linear vertical incision in the trachea (see Chapter 22, “Tracheostomy, Minitracheostomy, and Closure of Persistent Stoma”). The procedure is done in an operating room with aseptic technique. Bacteria are always present in the tracheal lumen and further colonization will occur after tube placement, despite exquisite postoperative care. Staphylococcus aureus and Pseudomonas aeruginosa are the most common. However, invasive sepsis may be limited by scrupulous postoperative care. The tracheostomy tube should be well seated and fastened securely to the patient’s neck. Avoidance of leverage on the tracheostomy tube is most important. The weight of connecting tubing and adapters, transmitted through the tracheostomy tube against the tracheal wall, causes erosion of the stomal margin. Long-term exposure to ventilation and other factors such as diabetes and corticosteroids are additional likely agents. Lightweight swivel adapters attached to the tracheostomy tube move in multiple planes and are connected by light, flexible corrugated tubing to the ventilator. It has been hypothesized that accordion tubing helps to avoid transmission of the respirator’s thrust to the stomal edges, and to the cuff itself, but conclusive experimental data is required. The ventilating connecting tubes are in turn suspended from supports. Clinical results support this protocol overall. Progressive careful attention to the points noted has eliminated stomal lesions at Massachusetts General Hospital.
Stenosis at Cuff Level Evidence pointing to pressure necrosis as the most important etiologic agent has been presented. Adriani and Phillips noted that variables such as cuff site, materials, and tracheal shape affected intracuff pressure.22 They found that intracuff pressure was not a direct index of pressure exerted on the tracheal wall. Knowlson and Bassett found that small increments over the minimal occlusive volume required to effect a seal in patients with a conventional endotracheal cuff, with 20 cm of water inspiratory pressure, caused a rapid rise in the pressure exerted on the tracheal mucosa.23 This exceeded arterial capillary pressure, especially against the anterior tracheal wall. Carroll and colleagues correlated intracuff pressures with pressures exerted on the tracheal wall by a variety of cuffs and found the relationship to be generally proportional.24 They set forth as criteria for ideal cuffs that these should have “large sealing areas, inflate evenly, and center the tube within the tracheal lumen;. . . . have large residual volumes requiring small additional volumes for ‘seal,’ low tracheal wall sealing pressure with overinflation.” Lomholt described a large-volume Teflon cuff with an attached trap, intended to maintain a constant cuff inflation pressure.25
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Cooper and Grillo proposed the use of a large-volume, low-pressure cuff which would conform to the irregular shape of the trachea when inflated, rather than establish a seal by expanding circumferentially and deforming an elliptical trachea to the shape of the cuff (Figure 11-16).15 The prototype of such a cuff was tested in experimental canine preparations. Conventional cuffs produced erosive and stenotic lesions (see Figure 11-13), whereas in equivalent time periods, the prototype large-volume, low-pressure cuffs resulted in no injuries other than slight submucosal inflammation (Figure 11-17). A latex cuff suitable for clinical use was designed, and when tested in humans for its sealing characteristics, it was found to require a tenth of the pressure required by a conventional high-pressure Rusch cuff.14 The cuff, roughly cylindrical in shape, measured approximately 2.4 cm in length and 3.0 cm in diameter when inflated to a pressure of 1 cm of water. At this point, the latex wall was unstretched, and the total volume of air that the cuff accepted prior to stretching was 12 cc (Figures 11-18A,B). The cross-sectional area of the filled but undistended cuff was greater than that of most adult tracheas, and could therefore fill out the configuration of the normal ovoid tracheal shape without applying stretch to the wall of the cuff itself. Initially, these cuffs were placed on conventional metal Jackson tracheostomy tubes, with care taken to prevent slippage. Randomized clinical trials following tests for safety compared the cuff’s performance in 25 patients, with 20 having standard cuffs.14 “Blind” endoscopic evaluation of damage to the tracheal wall on a scale of 0
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C 11-16 Diagram of a tracheal seal, attained by a high-pressure cuff versus a large-volume, low-pressure cuff. A, Cross section of the trachea and esophagus. B, The low-volume cuff is necessarily inflated to a high pressure to occlude the irregularly shaped tracheal lumen. C, The largevolume cuff expands to occlude the lumen, conforming to the shape of the lumen at low inflation pressure. FIGURE
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FIGURE 11-17 Experimental use of large-volume, low-pressure cuff in dogs. A, After 2 weeks of seal at the same ventilatory pressure used with high-pressure cuffs, only minimal mucosal inflammation results. Compare with Figures 11-13B,C. B, Experimental cuff, deflated.
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FIGURE 11-18 A, Rusch standard cuff (1971) at the left and experimental latex cuff on the right, mounted on Jackson metal tracheostomy tubes, both in a “resting” state at resting volume. The experimental cuff is deflated for insertion. B, Standard cuff (left) is inflated with 8 cc of air and has high intracuff pressure, and is asymmetric and quite rigid. The large-volume, low-pressure cuff is undistended with a similar volume of air, has no intracuff pressure, and is soft and symmetrical.
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to 4 was made immediately after deflation of the cuff as weaning began. Patients with the new cuffs had an average rating of 1.3 (median 1.0) in comparison with an average rating of 2.6 (median 2.5) for those with the standard cuffs. All patients in the minimum injury group had the new cuff in place. Few with the new cuff were in a category showing more serious damage. In contrast, the bulk of patients with the standard cuff fell into groups with progressively more severe damage, many lying in the ranges which were predictably likely to go on to clinical stenosis (Figure 11-19). The average intracuff pressure developed in the experimental cuff was 33 mm Hg compared to an average intracuff pressure of 270 mm Hg in the standard cuff. During the period of development of the cuff, following initial indicative experiments, Geffin and Pontoppidan proposed the interim use of prestretched Portex cuffs to approximate these conditions.26 Despite the limitations of this method, the incidence of cuff strictures in our respiratory care unit dropped noticeably with the prestretched cuffs, and totally disappeared following routine use of large-volume latex cuffs. Despite this clear enunciation of desirable standards for sealing cuffs for ventilation, followed by years of favorable experience, clinically available equipment still varies in characteristics. Latex is almost indefinitely extensible so that damaging pressures are not developed. However, the short shelf life of latex and the cost of attaching it to plastic tubes led to its abandonment. Large-volume cuffs are now generally available, but are made of relatively inextensible plastic materials. When the resting volume of the fully inflated, but unstretched, cuff is exceeded by only a few cc of overinflation, the lack of extensibility of the material leads to a rapid climb in intracuff pressure. The margin of safety is thus reduced with the relatively nonextensible material now used to fabricate cuffs. Ching and Nealon, and Ching and colleagues analyzed comparative characteristics of cuffs in several studies, confirming these findings (Figure 11-20).27,28 More extensible plastic would further improve safety. Large-volume cuffs should not be inflated beyond the minimum pressure that is adequate to provide ventilation without leakage. Personnel must also understand that cuffs have to be reinflated with care after routine deflation. Otherwise, the inflation volume, and consequently the pressure, creeps upward. It is principally the failure of proper management of cuff volume that continues to produce cuff stenoses today.
11-19 Damage to trachea from standard low-volume, high-pressure cuffs versus large-volume, low-pressure tracheostomy tube cuffs. Sixty-eight percent of patients with the experimental cuff showed no exposed cartilage (rating less than 2.0). No patient with standard cuff was in this category. Reproduced with permission from Grillo HC et al.14
FIGURE
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FIGURE 11-20
Comparison of intracuff and lateral tracheal wall pressures engendered by a then-standard Portex plastic cuff on the left and the newly developed latex large-volume cuff on the right. The occlusion volume for the trachea is indicated by an arrow in each case. If volume is exceeded, high intracuff and lateral tracheal wall pressures do not result in the large-volume cuff. (Illustration courtesy of Dr. NPH Ching and Dr. TF Nealon Jr.)
Seals other than air-filled balloons have been proposed, including flexible discs and a compressible synthetic sponge in an outer bag, which fills by expansion of the sponge matrix.29 A low-pressure pilot balloon was designed to relieve pressure in excess of a sealing level of 25 cm of water. 30 It was also proposed to reduce the time of exposure of an area of trachea to pressure, by alternate inflation of double cuffs in series. This proved to be unsatisfactory and, if anything, produced longer stenoses. Intermittent cuff inflation cycled to inspiration was also tried, with no instances of damage seen.31 None of these methods gained currency perhaps because of their relative complexity. Furthermore, they provide less protection against aspiration than sealing cuffs. Substitution of high-volume flow respirators has been suggested. Although it is possible to maintain children without sealing cuffs, adults with poor compliance and severe degrees of respiratory failure cannot currently be managed without a tracheal seal. Thus, we see that stomal strictures may be reduced to a minimum and perhaps eliminated. Information is available to eliminate cuff strictures. No cuff strictures have been produced at MGH since the initial introduction of a large-volume cuff. The problems that remain are the dissemination of information on management, coordination of manufactured equipment, and evolution of better materials. Prop-
Postintubation Stenosis
er use of large-volume, low-pressure cuffs and cessation of prolonged use of stiff nasogastric tubes should prevent tracheoesophageal fistulae (see Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula). Tracheo-innominate artery fistulae from cuff injury would also disappear. Those at the stomal level can be avoided by choosing the proper level for tracheostomy (see Chapter 13, “Tracheal Fistula to Brachiocephalic Artery”).
Clinical Presentation and Diagnosis Clinical Characteristics The majority of patients with postintubation tracheal lesions present clinically with obstruction. Principal manifestations are 1) progressive dyspnea, 2) wheezing and stridor, and 3) intermittent obstruction with retention of secretions. Pneumonitis or frank pneumonia may occur unilaterally or bilaterally. As the airway narrows, dyspnea on effort is noted first. This appears initially with marked effort, depending on the respiratory reserve of the patient. In time, dyspnea appears with less exertion. Many patients with benign tracheal stenosis remain sedentary or bedridden for a long time due to their original illnesses. Severe degrees of obstruction may therefore occur before clinical symptoms become obvious. In a patient on bed rest, the airway may contract to a diameter of 5 or 6 mm before symptoms are recognized. Other patients with a severe but fixed stenosis that is no longer progressing are dyspneic, only as they become more active during recovery from illnesses such as polyneuritis. Slow progression of stenosis may lessen a patient’s awareness that a change in airway function is occurring. In most cases, however, the rate of closure is relatively swift. Sometimes, symptoms follow immediately or within days after removal of a tracheostomy tube. Obstruction may also occur from granulation tissue while the patient is still tracheostomized. With the most severe degrees of airway obstruction, the patient may be unable to lie down or complete a sentence without gasping for breath. As the airway narrows, wheezing occurs, followed by frank stridor. Classically, an upper tracheal obstruction outside of the thorax will present with severe inspiratory stridor, and a low intrathoracic stenosis with expiratory wheezing. Usually, stridor may be produced in either phase on deep breathing with effort. Later, the wheeze is present at rest. A marked inspiratory high-pitched sound may be heard across the room even when the patient is quietly seated. When stridor becomes audible at rest, a high degree of obstruction is usually present, with the airway measuring less than 6 or 7 mm in diameter. At this point, action is urgently demanded. The stridor is elicited by having the patient breathe in slowly and deeply through an open mouth, and then forcing the breath out rapidly, with mouth still open. An attempt to inspire deeply and suddenly will often lead to severe coughing. Auscultation over the trachea and upper chest will further identify stridor. It may be heightened by the forced expiratory maneuver described. Although some of these sounds are transmitted peripherally, they are more remote on auscultation over the peripheral lung fields. In contrast, wheezing due to asthma and bronchitis is peripheral and is not heard maximally over the trachea itself. As the airway narrows it becomes increasingly difficult to clear secretions. Plugs of mucous accumulate, occasioning transient episodes of worsened obstruction. The patient may cough violently in an effort to clear the airway, becoming plethoric and then cyanotic. Episodes of transient obstruction usually signal a marked degree of airway obstruction with an aperture that may measure less than 5 mm in diameter. The fact that such an episode may be cleared with chest physiotherapy or suctioning does not lessen the gravity of the warning. A subsequent obstructive episode may well be fatal. Pneumonitis and pneumonia occur in the presence of tracheal obstruction. Most commonly, however, the lung fields remain clear on the x-ray. This is the reason why so many patients with severe obstructive tracheal lesions fail to be diagnosed promptly. Assumption is made that the disease must be bronchi-
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tis or asthma, and too many patients have been treated over long periods for “adult onset asthma,” with an illusion of response. Some have been placed on high doses of prednisone prior to referral. The basic diagnostic rule to be remembered is that any patient who presents with dyspnea on effort, wheezing, or episodes of airway obstruction, and who has been intubated and ventilated at any time in the recent past, must be considered to have organic upper airway obstruction until proved otherwise. The corollary should be added that if a history of intubation is lacking, tumor or other obstructing disease of the upper airway should be excluded. With this rule in mind, diagnosis is not difficult, especially with the ready availability of the flexible bronchoscope. Analysis of a group of our early patients with stenosis following tracheostomy for respiratory care showed an equal gender distribution and a mean age of 47 years (range 16 to 79 years). The causes of the original respiratory failure were diverse, including chest trauma, drug ingestion, myasthenia gravis, polyneuritis, head injury, pickwickian syndrome, pneumonia, and following cardiac surgery. Tracheostomy had been required in these patients for periods ranging from 2 to 119 days, with a mean duration of 42 days. Several different types of tracheostomy tubes had been used in this group of patients, but metal tubes (silver or stainless steel) with rubber cuffs, or plastic tubes with plastic cuffs, predominated in this era prior to the development of low-pressure cuffs. In many cases, at least two kinds of tube and cuff had been used. In most, an endotracheal tube had been employed for a period up to 4 or 5 days prior to tracheostomy. In 3 patients, the inflatable cuffs had been used without ventilator assistance to prevent aspiration of foreign material into the lungs. In the remaining patients, the cuffs were used in association with artificial ventilation. Ventilation was administered for 3 to 112 days with a mean duration of 33 days. Although many of the patients had been treated at other hospitals prior to referral for correction of their stenosis, the care of patients who developed stenosis at the Massachusetts General Hospital included hourly deflation and reinflation with no more air than was required to effect a seal at peak airway pressure. All procedures and suctioning were done with aseptic precautions. In patients in whom precise information was available on the time interval between extubation and the onset of symptoms, it was found that 18 had symptoms within 30 days and 24 had symptoms within 90 days of extubation. One patient was seen 18 months following extubation. In some patients, the symptoms were evident within a few days after removal of the tube. A 30- to 90-day interval, however, meant that many of these patients were discharged from the hospital prior to development of symptoms. This undoubtedly accounts for the fact that so many were treated for “adult onset asthma” or other vague diagnoses. Clinical history is a most important element in diagnosis. A later analysis of 156 postintubation lesions treated in the decade between 1965 and 1975 showed that 14 patients had never had tracheostomy, but had developed stenosis from the cuff on endotracheal tubes. Several had been intubated for periods less than 48 hours and one case was for less than 36 hours. Of the lesions related to tracheostomy tubes, 72 were due to cuff stenoses, 51 to stomal strictures, 9 had had both lesions present, and the etiology was uncertain in 1 other case. Two patients had segments that demonstrated malacia only; both were located in areas of cuff damage. There were 6 tracheoesophageal fistulae due to cuffs and 1 fistula to the brachiocephalic artery due to a cuff erosion. A review carried out in 1995 showed an increasing ratio of stomal over cuff lesions in patients with tracheostomy tubes (from 1:1.4 in the decade of 1965–1975 to 2.3:1 in the two decades from 1975–1995), probably reflecting the introduction of low-pressure cuffs and their correct usage. 32 Increasing use of endotracheal tube ventilation is suggested by an increasing ratio of cuff stenoses from endotracheal tubes over those from tracheostomy tubes (1:5.1 in 1965–1975 compared to 1.8:1 in 1975–1995). Increased incidence in laryngotracheal subglottic stenosis, chiefly the result of ventilation with endotracheal tubes, also reflects this changing preference in chosen route of administration of mechanical ventilation.
Postintubation Stenosis
Diagnosis When the presence of stenosis is suspected on the basis of the history, symptoms, and signs, appropriate imaging studies will quickly define the location and extent of the lesion. Once a patient begins to have stridor and shortness of breath on minimal exertion, the lesion may progress rapidly toward complete obstruction. A small plug of mucous or edema may close the airway completely. Such patients must be hospitalized at once, watched carefully in a respiratory care unit, and studies completed urgently. Conventional radiologic images are often more useful than a computed tomography (CT) scan or CT-derived reconstructed images (see Chapter 4, “Imaging the Larynx and Trachea”) (Figures 11-21A–E). Contrast medium is not necessary and may cause some difficulty in patients who have high degrees of obstruction. Fluoroscopy is essential as an added means of assessing glottic function and to detect tracheomalacia (see Figures 11-14A,B). It is extremely important to define all tracheal lesions and to analyze the status of the larynx precisely, since concurrent lesions do occur. The treatment of specific lesions varies and a total plan should be based on complete information. In particular, it is important that an effective functional laryngeal airway be assured before tracheal reconstruction is undertaken. It may be necessary to temporize even with a tracheostomy while an affected larynx is initially repaired. Synchronous repair of the larynx and trachea is possible but can add risk.33 High tracheal lesions must be differentiated from those that also involve the subglottic larynx.8 An early surgical failure in our series was due to failure to recognize a significant degree of proximal malacia in addition to stenosis. Another failure was due to a lack of appreciation of glottic inadequacy, in a patient with a complex tracheal stenosis. Informative tracheal x-rays should be available prior to endoscopy. Findings on bronchoscopy may be somewhat confusing to a surgeon who has not seen many of these lesions. X-rays serve as a road map for the bronchoscopist. Also, malacia may not be recognized bronchoscopically, especially under general anesthesia, without prior warning of its presence from fluoroscopic examination. Diagnostic bronchoscopy is discussed in Chapter 5, “Diagnostic Endoscopy.” It is my firm belief that every patient with a diagnosis of “adult onset asthma” should be examined bronchoscopically to rule out organic obstruction.
Management and Results Urgent Management A number of patients will have been treated previously for acute respiratory arrest in a hospital from which they were referred. Others will arrive with high degrees of obstruction in very tenuous status. Respiratory decompensation can occur very soon after admission. If a hospitalized patient develops nearly complete obstruction of the airway after failure to recognize the severity of the problem, or suddenly deteriorates while under observation, emergency endotracheal intubation may be required. No effort should be made to push a tube through the stenosis. Rather, the endotracheal tube should be placed above the stricture and the airway suctioned. With positive pressure ventilation, it is almost always possible to maintain the patient with a Venturi-like flow through the stenosis. A laryngeal mask airway can be used for a subglottic stenosis where proximal intubation is not possible. The patient should be moved promptly to the operating room, where a rigid bronchoscopy and dilation of the stenosis can be done under general anesthesia without respiratory paralysis. Very few patients, even those in borderline state, require such urgent intubation. Immediately upon arrival to the hospital, the patient should be placed in a high-level intensive care facility, preferably a respiratory unit, where intubation can be done at a moment’s notice and where there is constant attendance by appropriately trained physicians. With gentle physiotherapy, suctioning, adequate humidification, and supplemental oxygen or heliox, and with light medication to control anxiety, the patient usually settles down and is quite comfortable. The time gained may be used for obtaining appropriate diagnostic x-rays and ini-
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B
A
C
FIGURE 11-21 Roentgenographic delineation of postintubation stenosis. A, Filtered view of entire upper airway, in a 60-year-old man with recurrent upper tracheal stenosis after prior resection of the stomal stenosis elsewhere. Stenosis followed ventilation via tracheostomy for respiratory failure after coronary artery surgery. Easily identifiable are the false vocal cords, glottis (arrow), subglottic larynx, stenosis (arrow), and the length of normal trachea remaining. B, Evolution of a cuff stenosis for ventilation with an endotracheal tube only. On the left is a detail from a chest x-ray showing the endotracheal tube and the distended balloon cuff (arrows). On the right is the stenosis (arrows) in the same tracheal segment, from another plain chest x-ray. C, The stenotic lesion from B shown on a computed tomography scan. The left image shows the beginning of the stenosis whereas the right image shows the middle of the lesion. Note the nearly complete destruction of the cartilage, and the massive scar tissue contracting the lumen to a fraction of normal cross section.
tial clarification of the patient’s medical condition. An elective corrective surgical procedure may be planned and performed under ideal circ*mstances. This will minimize errors in appraisal of the lesion and of the larynx. If the patient’s condition fails to improve or deteriorates, the surgical team should move promptly to bronchoscopic evaluation and dilation under general anesthesia. A technique for safe emergency tracheal dilation is detailed in Chapter 19, “Urgent Treatment of Tracheal Obstruction.” Dilation is also a method of temporizing while a patient is further evaluated and
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D
E FIGURE 11-21 (CONTINUED)
D, Tomograms outlining areas of stomal stenosis and anastomosis pre- and postoperatively (left and right images, respectively). Note the irregularity of the right side of the subglottic larynx preoperatively. There is slight narrowing at the anastomosis. E, Computed tomography scans comparing a nearly normal trachea at left image with side-to-side narrowing of a stomal stenosis on the right image. Indentation of the cutaneous scar is seen. At bronchoscopy and operation, the anterior cricoid cartilage proved to be completely eroded, and thus required laryngotracheal resection and reconstruction. Imaging and endoscopy are both essential before an operation.
medical or other surgical problems are corrected. Dilation may be ineffective in a patient with stomal stenosis, but such patients usually do not obstruct so totally. In circumferential cuff stenosis, dilation is essentially always only transiently effective. In most patients, dilation is effective for days to weeks or longer. Tra-
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cheostomy is best avoided if it appears possible to move ahead soon to a definitive surgical treatment. A fresh tracheostomy usually delays surgical repair and, worse, may damage normal trachea necessary for reconstruction, if the tracheostomy is mistakenly placed other than through the lesion itself. Dilation, performed carefully by an experienced endoscopist, is also a procedure that may be used in an institution where there is little experience in tracheal reconstruction. A patient may then be transferred safely to a center where such work is done regularly, without the delaying or potentially damaging effect of tracheostomy. Transfer should be expeditious since the duration of relief from dilation is unpredictable. A small endotracheal tube may be inserted through the dilated lesion, if necessary, for transportation. Dilation through an existing stoma is described in Chapter 19, “Urgent Treatment of Tracheal Obstruction.” There is little practical use for lasers in these situations. Some improvement may be obtained by lasering away small amounts of granulation tissue or scar, but this should be no more than is usually obtained by dilation with, sometimes, use of bronchoscopic biopsy forceps. Radial lasering and dilation seem to have little benefit greater than dilation alone. It must be recalled that the pathology of a tracheal stenosis most often involves hourglass or side-to-side narrowing of the trachea, so that aggressive destruction of tracheal wall can result in perforation. If for any reason surgical treatment is not elected, or if the patient is nontransportable, a tracheostomy may be necessary for longer-term management, even if not as the definitive treatment. Bronchoscopic dilation is done first under general anesthesia. The rigid ventilating bronchoscope used for dilation should remain in place for ventilation and as a guide for tracheostomy. If there was a prior tracheostomy, it is often advisable to reinstitute the tracheostomy at precisely the same point where the previous opening was made, especially if it lies in the stenotic segment (Figure 11-22A). If the stricture is stomal, no additional trachea will be damaged by a second tracheostomy. A cuff stenosis that lies in the cervical trachea is the best site for a necessary tracheostomy (Figure 11-22B). If the lesion is a cuff stenosis and lies at or below the sternal notch, then the least damage will be done, with few exceptions, if the old tracheostomy site is carefully reopened (Figure 11-22C). The procedure is minimal since the scar leads directly to the trachea. A transverse incision about 1 cm in length in the prior tracheostomy scar will usually suffice. The tracheostomy tube must be passed through the stenosis, confirmed by flexible bronchoscopy through the tracheostomy tube. Otherwise, a false sense of security will be obtained while the stenosis may close down below the tip of the tracheostomy tube. If a prior surgeon had recently placed a new tracheostomy inferior to an obstructing stenosis, which threatens to increase the extent of tracheal damage, then this length of potentially useful trachea may be recaptured, by replacing a tracheostomy through the stenotic segment and permitting the new, inferior stoma to heal (Figure 11-22D). If operation must be delayed a T tube provides a patient with a more normal airway and with voice. Dilation of a tracheal stenosis, reinstitution of a tracheostomy at an appropriate level, and reinsertion of a tube of proper length or a T tube is also a method of permanent management of a tracheal stenosis where repair is contraindicated. If early reconstruction is envisaged, peroral dilation is the method of choice. Rarely would a silicone stent be considered as a bridge to later resection, and never should an expandable stent be considered at all. Postintubation tracheoesophageal fistula and tracheo-innominate fistula are discussed in Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula,” and Chapter 13, “Tracheal Fistula to Brachiocephalic Artery.”
Selection of Patients for Reconstruction Most patients with stenotic lesions of the trachea can be managed indefinitely by reinstituting a tracheostomy and placing an appropriate splinting tube, such as a Montgomery silastic T tube, through the stenosis. This places a great burden on a surgeon proposing reconstruction, who must then confidently be able to offer a very high chance of success for a proposed surgical alternative to a T tube. This is heightened by the fact that
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FIGURE 11-22 Correct placement of tracheostomy tubes, when necessary, to manage postintubation tracheal stenoses. A, A stomal stenosis is best treated by locating the new tracheostomy in the stenotic segment, never below it. B, A high cervical cuff stenosis is best treated by locating the new tracheostomy in the stenotic segment, never below it. C, A low substernal cuff stenosis is managed by tracheostomy in a prior cervical site or at the conventional (2d–3d ring) site, with placement of a tube long enough to extend through the distal stenosis. D, If a cervical stenosis of considerable length has been managed by tracheostomy placed distal to it, the tracheostomy site should be relocated to the stenosed segment. The inferior tracheostomy is allowed to heal, thus recapturing a usable distal tracheal length. T tubes are also placed in the same locations in each case for longerterm management of patients, providing voice and more normal respiration.
the best opportunity for successful reconstruction lies in the initial surgical attempt. Second trials may or may not succeed and a third attempt entails even more risk. Tracheal reconstructive procedures should not be undertaken without considerable study and experience. Silicone or expandable stents, even if coated, appear inadvisable to treat benign stenosis, since they not only produce severe stenotic lesions but may make future definitive repair impossible (see Chapter 40, “Tracheal and Bronchial Stenting”).34
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As techniques evolved and experience increased, I rejected few patients for “medical reasons,” particularly those with poor respiratory reserve or marginal cardiac status. Essentially, all postintubation tracheal stenoses may be repaired through an anterior approach, avoiding entry into the pleura or pericardium, even with lesions at the supracarinal level (see Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection”). With skilled anesthesia and with exquisite intra- and postoperative management of the airway, few patients need to be denied curative treatment. The patient who remains in need of a respirator, however, should not be considered for surgical reconstruction unless there is absolutely no alternative way to maintain the airway, a situation which rarely pertains. A patient who will predictably require prolonged mechanical respiratory support postoperatively is also best deferred as a surgical candidate. Even a low-pressure cuff in contact with a fresh anastomosis for a long period of time will incite inflammation, which may lead to dehiscence and death. Following extensive tracheal resection, it may be impossible to seat a cuff that will not impinge on the anastomosis in the shortened trachea. I have preferred not to perform reconstructions in patients who have basic diseases that will almost certainly require another tracheostomy within a very short period of time. An example of this is a patient who suffers from severe myasthenia gravis and has required multiple tracheostomies over the course of illness. For such a patient, a fenestrated tracheostomy tube or a silastic T tube seems preferable to provide airway and speech. I do consider patients with a borderline pulmonary status for reconstruction. In these patients, a calculated risk is taken, after full discussion with the patient, with the understanding that should another attack of respiratory failure in the future occur, then another tracheostomy, temporary or permanent tracheostomy may be required. Thus far, such patients have been satisfied to obtain a prolonged or, most often, indefinite relief from tracheostomy. With proper consultation and management, stable coronary artery disease is not a contraindication to reconstruction. Proponents of lasers and stents have all too readily accepted these conditions as reasons to deny patients definitive surgical treatment. Elective tracheal reconstruction is best deferred in patients who are on chronic high-dose corticosteroid therapy. Although healing does occur slowly in the presence of significant doses of steroids, the chance of dehiscence is increased. Even with only moderately long resection, and hence moderate anastomotic tension, the result may be slow distraction due to delayed collagenous healing, slow increase in tensile strength, and subsequent restenosis. In 2 patients who suffered anastomotic stenosis following reconstruction, while on about 50 mg of prednisone daily, a successful re-resection was later accomplished when they were no longer steroid dependent. Where possible, it is preferable to wean the patient from the drug completely or to low doses before a tracheal operation. In the interim, the patient may be carried by repeated dilations, or with a T tube, if dilation is required too frequently. A paradoxical problem arises in a patient whose myasthenia gravis is successfully controlled with chronic steroid treatment, but who has a tracheal stenosis. Here, conservative management with a T tube may be preferable. Demonstrated subtotal destruction of the trachea contraindicates reconstruction. An example is a patient who has only 2 or 3 cm of adequate trachea remaining. In postintubation lesions, this is almost always a result of inappropriate attempts at surgical repair of the trachea, and almost never from the original lesion. Although it may be possible to try to rebuild such tracheae in stages or to attempt replacement with a prosthesis, the first method is complex and the second is experimental. Failure is frequent and may be fatal (see Chapter 45, “Tracheal Replacement”). A safe and relatively satisfactory alternative is to splint the trachea with a Montgomery silastic T tube (see Chapter 39, “Tracheal T Tubes”). Pearson and Andrews preferred to defer operation until a fresh stenosis had “matured” and acute inflammatory changes had subsided.35 In the presence of florid granulations and acute inflammation, I completely agree that it is judicious to delay surgery until the inflammatory reaction subsides. This may require weeks, months, or longer. Following a prior failed attempt at reconstruction, at least 4 months, and preferably 6, should pass before a second operation. Even then, surgical planes will be difficult and the chance of success somewhat diminished.
Postintubation Stenosis
A variety of treatments have been used for postintubation stenosis, including repetitive dilation with or without injection of steroids into the area of inflammation, lasering, or, usually more effectively, the insertion of conventional internal splints (a tracheostomy tube or silastic T tube). Rarely, a patient with a lesion characterized by incomplete destruction of the tracheal wall in depth, extent, or circumference, may achieve a permanently open airway after a prolonged period of stenting. Lesions that respond to such treatment are usually of lesser severity. As the inflammatory process subsides and the scar becomes more mature, a partial airway is obtained. In order for this to occur in the trachea, it must be assumed that part of the structural integrity of the cartilaginous wall remained. Treatment requires a prolonged period of time and often results in a less than ideal airway. When the splinting tube is removed, the patient must remain under close observation, since in most cases, the stenosis will contract again over time and ultimately require surgical reconstruction. Stenting should not be a routine step in treating tracheal stenosis, since success by this method is so rare. Furthermore, expandable stents, even when coated, may cause severe proximal and distal granulation and stenosis, extending the original lesion to unresectable lengths (Figures 29 and 30 [Color Plate 15]).34 Such stents should not be used, especially where the lesion is a correctable one. In most patients, indications for surgical correction are evident when the patient is first seen. For most patients, there is almost no hope for success of a “conservative” treatment. The alternatives are a permanent tube or surgical reconstruction. Patients who have had splinting tubes in place for months, or even years, will usually close their airway acutely in the 40 or 50 minutes required for tracheal radiography to be completed with a tracheostomy tube removed. Following removal of a T tube, distress usually occurs in days to weeks. As surgical techniques and experience with tracheal reconstruction grew, I felt increasingly justified in advising early reconstruction rather than tedious trials of uncomfortable conservative treatment, which could rarely succeed. Repair has only been deferred in the special categories described. Especially to be avoided are inlying stents, which make lesions surgically incurable. Even silicone stents for lesions high in the trachea may incite granulation tissue if they impinge on the conus elasticus of the subglottic larynx.
Definitive Treatment Surgical techniques, applicable to correction of postintubation stenosis are detailed in Chapter 24, “Tracheal Reconstruction: Anterior Approach and Extended Resection,” and Chapter 25, “Laryngotracheal Reconstruction.” The surgical management of postintubation tracheoesophageal and tracheoarterial fistulae is related in Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula,” and Chapter 27, “Repair of Tracheobrachiocephalic Artery Fistula.” The rare lesion of isolated malacia resulting from intubation is best treated by segmental resection, where its length is not excessive.
Results of Surgical Treatment Patients. In the 27 years between 1965 and 1992, 503 patients underwent tracheal resection and reconstruction for postintubation lesions at Massachusetts General Hospital.32 There were 266 males and 237 females with an age range of 6 to 85 years (average 44 years). Of these, 251 lesions resulted from the sealing cuff of an endotracheal or tracheostomy tube, 178 were at the site of a tracheostomy, and 38 had evidence of both lesions. In 36 cases, the exact site of origin was not certain, often because of previous attempts at treatment, including multiple tracheostomies. In 441 patients, the lesions were principally tracheal. Sixty-two had involvement of the subglottic larynx as well as the upper trachea. In 123 patients, ventilation had been provided only with an endotracheal tube. Most patients had received ventilatory assistance initially via an endotracheal tube for varying periods of time before tracheostomy, and 380 had either already had a tracheostomy or were given one when seen. Of note is the fact that 2 patients had been intubated for less than 18 hours, several for 48 hours or less. Nearly all had received ventilatory assistance.
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Many patients had undergone prior attempts at surgical treatment before referral. These included resection (53), tracheal operations such as wedge resections, splinting, or fissure (31), and laryngeal procedures such as stenting, grafting, or fissure (20). Sixty had had T tubes placed and at least 45 had undergone laser treatment. Eight patients had had prior repairs of tracheoesophageal fistula, 3 of which had failed. Operative Treatment. At MGH, 521 tracheal resections were done on the 503 patients described above. Thirteen of them who had restenosis after initial operation were later reoperated upon. Five patients with immediate failure because of residual malacia were reoperated on within hours of the initial operation. These cases are not counted as additional reconstructions but as complications. Since this series includes our entire experience, the surgical technique had evolved over the 27 years spanned. Surgical procedures are described in Part 2 of this book. Approach was through a cervical incision in 350 patients, through a partial upper sternotomy to a point below the sternal angle (cervicomediastinal) in 145 patients, with 2 additional patients requiring extension of the cervicomediastinal incision into right anterior thoracotomy. Earlier, a right posterolateral thoracotomy was used for 6 patients. Few if any reconstructions for postintubation stenosis would now be done through thoracotomy. A cervical or upper cervicomediastinal incision suffices, even for lesions at the supracarinal level. Complete sternotomy is unnecessary. The amount of trachea resected most commonly measured between 2 to 4 cm with a range from 1 to 7.5 cm. Anastomotic tension was routinely lessened by complete dissection of the pretracheal plane and by cervical flexion at the time of anastomosis. Laryngeal release was used in 49 patients, the first 9 by the thyrohyoid technique of Dedo and Fishman,36 which Dedo has now abandoned, and the remaining 40 by the suprahyoid technique of Montgomery.37 Laryngeal release was used in 9.7% of patients. Only 8% of the 450 patients who had not undergone prior tracheal resection were thought to require laryngeal release to reduce anastomotic tension, in comparison with 24% of the 53 who had had prior resection and reconstruction. In only one complex case, an intrapericardial hilar release was added. Trachea-to-trachea anastomosis was performed in 324 patients, trachea-to-cricoid anastomosis (with horizontal removal of varying amounts of the anterior cricoid cartilage) in 117 patients, and laryngotracheal anastomosis (with removal of the anterior cricoid arch of the subglottic larynx) in 62. Laryngeal release was used in 29 of 324 (9%) patients with trachea-to-trachea anastomosis, in 12 of 117 (10.3%) with trachea-to-cricoid anastomosis, and in 8 of 62 (12.9%) with trachea-to-thyroid cartilage anastomosis. The use of laryngeal release was dictated by the extent of resection and tracheal mobility in each patient. It was necessary more often in patients who had undergone prior resection or in whom the lesion extended into the lower larynx. Laryngeal release should not be employed routinely. Although the technique of placement of sutures varied little over the period described, suture material progressively changed. Prior to 1978, 4-0 Dacron polyester, Tevdek polyester, Mersilene polyester, and Prolene polypropylene sutures were used in a search for improvement. Sufficiently fine, absorbable catgut was not strong enough for use. Wire presented a threat to an adjacent brachiocephalic artery. Since 1978, Vicryl polyglactin 910 has been used. The change was dictated by the frequency of granulomas at the suture line with all nonabsorbable sutures listed. Granulations essentially vanished following change to the use of absorbable Vicryl. Suture line granulations dropped from an incidence of 23.6 to 1.6%, and most are now not of clinical importance. Monofilament PDS polydioxanone was tried and discarded since no advantage over Vicryl was found and it was somewhat more difficult to use. Twenty-five patients in the series had tracheoesophageal fistulae as well. Seven patients with stenosis accompanied by extensive tracheomalacia were managed with the placement of one or more polypropylene rings around the malacic segment of the trachea, usually above the level of the tracheal resection for stenosis. In 84 patients, the anastomoses were covered with adjacent tissues: thyroid isthmus in 50, cervical strap muscle in 26, and other tissue including thymus and pericardial fat pad in 8. If the pathology and, therefore, the anastomosis lay adjacent to the innominate artery, or if the artery had been dissected in a
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prior operation, then the tissue (usually pedicled sternohyoid muscle) was interposed between the anastomosis and the artery. Routine wrapping of the anastomoses was not deemed necessary in the cervical or mediastinal region with these approaches, in contrast to intrathoracic anastomoses. Results. In the MGH series, results were classified as good, satisfactory, failure, and death. Good indicates a patient being functionally able to perform usual activities, with an anatomically, essentially normal airway, as determined by postoperative roentgenograms or bronchoscopy. Results are considered satisfactory if the patient can perform normal activities but is stressed on exercise, or if there exists an abnormality such as a paralyzed or paretic vocal cord, or where significant airway narrowing is evident on endoscopy or roentgenograms, even if the patient’s level of activity does not clinically evidence this. Patients were considered failures if they required a permanent tracheostomy or T tube to maintain an airway. The average length of follow-up was 3 years. In our experience, patients who remain stable after 2 months are very unlikely to have further difficulties. By 6 months to a year, the result may be considered to be final. Later change in clinical status was not observed. Results were good in 440 patients and satisfactory in 31 patients, and there were 20 failures and 12 deaths (Table 11-1). Of the failed patients, 11 were treated with a tracheostomy, 7 with a T tube, and dilations in 2. Factors in the Results. Prior resection and reconstruction led to 72.2% good results in comparison with 85% good results in those without prior resection and reconstruction (see Table 11-1).32 Combined good and satisfactory results were 90.2% and 94.4%, respectively, with and without prior resection. The failure rate of the first operation was 3.8% with a 2.4% mortality, compared with 5.6% failure with a prior resection. Despite a surprisingly high proportion of good results (87%), reconstruction after prior complex tracheal operations such as insertion of a Marlex prosthesis, cartilage grafts, hyoid bone grafts, cutaneous trough operations, and stented cutaneous grafts, led to the highest failure rate (9.7%) (Table 11-2). A previous T tube, laser therapy, or prior repair of tracheoesophageal fistula did not seem to affect outcome adversely. The failure rate increased with a higher level of anastomosis (Table 11-3): trachea-to-trachea anastomosis 2.2%, trachea-to-cricoid 6.0%, and trachea-to-thyroid cartilage 8.1%. Minor complications became more prevalent with each level (from 16% to 17.1% to 21%) but major complications were unchanged (13.9%, 15.4%, 12.9%, respectively). Laryngeal release was performed in 46 patients with an average length of resection of 4.4 cm. Forty-one were done at the initial operation and 5 at reoperation. Three additional patients had release before referral. Of the 41 released initially, results were good in 77%, satisfactory in 9.1%, and failure or death occurred in 6.8%. The 5 who underwent release with reoperation and the 3 with a prior release all had good outcomes. Of the 20 patients with tracheoesophageal fistulae also, including 3 with failed prior repairs, 18 had a good outcome, 1 was satisfactory with reoperation, and 1 died. Patients with short segments of tracheomalacia were treated by resection. However, 7 who underwent tracheal resection for stenosis had extensive
Table 11-1 Results of Surgical Treatment of Postintubation Tracheal Stenosis
Initial operation Reoperation Overall
Good
Satisfactory
Failure
Death
Reoperation
Number of Patients
Number
%
Number
%
Number
%
Number
%
Number
%
503 18 503
427 13 440
84.9 72.2 87.5
27 4 31
5.3 22.2 6.2
19 1 20
3.8 5.6 3.9
12 0 12
2.4 0 2.4
18 0 0
3.6 0 0
Reprinted with permission from Grillo HC et al.32
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Table 11-2 Effect of Prior Treatment on Results of Surgical Treatment of Tracheal Stenosis Good Treatment T tube Laser Resection and reconstruction Other tracheal surgery Laryngeal surgery TEF repair No prior treatment No prior resection and reconstruction
Satisfactory
Failure
Death
Reoperation
Total
Number
%
Number
%
Number
%
Number
%
Number
%
60 45 53
51 40 40
85 88.9 75.5
0 1 6
0 2.2 11.3
3 1 3
5 2.2 5.6
2 0 2
3.3 0 3.8
4 3 2
6.7 6.7 3.8
31
27
87.1
1
3.2
3
9.7
20 8 342 450
16 7 295 387
80 87.5 86.2 86
2 0 18 21
10 0 5.3 4.7
0 0 12 16
0 0 3.5 3.6
1 0 8 10
1 1 9 16
5 12.5 2.7 3.6
5 0 2.3 2.1
Reprinted with permission from Grillo HC et al.32 TEF = tracheoesophageal fistula.
additional malacic segments splinted with polypropylene rings. Three had a good outcome, 1 failure required a permanent T tube, and 1 died. Two reoperations resulted in 1 good and 1 satisfactory result. This procedure is not a standard operation, is not recommended, and requires further study and development. The danger points are potential loss of blood supply to the malacic segment and infection around the foreign material in a procedure where the trachea is also opened. Postoperative reintubation was necessary in 23 patients: 9 on the day of surgery, 11 in the first postoperative weeks, and 1 at 30 days. Proximity of repair to the glottis increased the likelihood: 9 reintubations in 324 trachea-to-trachea, 8 in 117 trachea-to-cricoid, and 6 in 62 trachea-to-thyroid cartilage anastomoses. Since reintubation indicated a major problem, it is not surprising that 4 of these patients needed permanent tracheostomy or T tube and that 5 patients died. In 27 patients, a concurrent tracheostomy was deemed advisable at completion of the reconstruction for factors such as laryngeal edema, vocal cord paralysis, or severe and uncorrectable narrowing immediately below the glottis. Tracheostomy was initially routinely performed in our early series of laryngotracheal reconstructions but is now used only occasionally for specific indications. Four T tubes were also placed, for such reasons as control of a high second stenosis after resection of a lower one. Twenty-one obtained
Table 11-3 Effect of Level of Anastomosis, Presence of TEF, and Use of Ring Supports on Results of Surgical Treatment of Tracheal Stenosis Good Treatment Anastomosis Trachea-trachea Trachea-cricoid Trachea-thyroid TEF repair Plastic ring supports used
Satisfactory
Failure
Death
Reoperation
Total
Number
%
Number
%
Number
%
Number
%
Number
324 117 62 20 7
275 101 51 18 3
85.9 86.3 82.2 90.0 42.9
18 4 5 0 0
5.5 3.4 8.1 0 0
7 7 5 0 1
2.2 6.0 8.1 0 14.3
9 2 1 1 1
2.8 1.7 1.6 5.0 14.3
15 3 0 1 2
Reprinted with permission from Grillo HC et al.32 TEF = tracheoesophageal fistula.
%
4.6 2.6 0 5.0 28.5
Postintubation Stenosis
good results, 3 satisfactory, whereas 1 required reoperation and 2 required permanent tracheostomy. The placement of a complementary tracheostomy and its compartmentalization from the fresh anastomosis and from the innominate artery are detailed in Chapter 25, “Laryngotracheal Reconstruction.” Complications of operations are summarized in Table 11-4 and discussed in detail in Chapter 21, “Complications of Tracheal Reconstruction.” Deaths occurred in 12 patients perioperatively. Anastomotic dehiscence and its complications accounted for 7 deaths, including 2 with consequent innominate artery hemorrhage. One of these had undergone a resection through massive fibrosis of prior irradiated Hodgkin’s disease, and reconstruction failed despite omentoplasty. Retrospectively, healing was not possible. Subsequent patients with this lesion have been managed with T tubes. Early in our series, 2 patients with previously failed resections elsewhere were reoperated on, while on respirators, a situation that should be avoided. Reintubation for secretions and flail chest accounted for the other dehiscences. In the era when only high-pressure cuffs were available, reintubation after extensive resection soon led to dehiscence. Low-pressure cuffs resting on a fresh anastomosis also encourage separation, but after a longer interval of irritation. If ventilation is needed after tracheal reconstruction, it is best to site the cuff above or below the anastomosis, if possible. One patient died from a postoperative innominate artery fistula, 2 from malacic tracheal obstruction, 1 from myocardial infarction, and 1 from respiratory failure late at home without clarification of cause.
Reconstruction after an Unsuccessful Repair Reconstruction following a prior attempt is daunting because of reduced length of normal trachea available, and surgical scar, which makes dissection difficult, endangers recurrent laryngeal nerves, limits tracheal mobility, and may affect tracheal blood supply. Reoperation should be delayed for 4 to 6 months after a prior reconstruction to allow subsidence of tissue inflammation and edema, and maturation of scar. We
Table 11-4 Complications of Operations for Postintubation Tracheal Stenosis
Granulations Before 1978 After 1978 Dehiscence Laryngeal dysfunction Malacia Hemorrhage Edema (anastomosis) Infection Wound Pulmonary Myocardial infarction Tracheoesophageal fistula Pneumothorax Line infection Atrial fibrillation Deep venous thrombosis Total Reprinted with permission from Grillo HC et al.32
Major
Minor
Total
11
38
49
10 1 28 11 10 5 3 12
34 4 1 14 0 0 1 22
7 5
44 5 29 25 10 5 4 34
8 14
15 19
1 1 0 0 0 0
0 0 3 1 1 1
1 1 3 1 1 1
82
82
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reoperated on 75 patients, 16 failures of our own and 59 referred after failure elsewhere.38 They had been managed with observation, dilation, tracheostomy, or T tube. Nineteen required laryngeal release and complications were more frequent. Results were surprisingly good in this group: 78.6% were good, 13.3% satisfactory, and 5.3% were failures. Two died (2.75%) after dehiscence. Failure of tracheal reconstruction is best treated with a permanent T tube if there is insufficient tissue for a safe reconstruction. Although 16 of our own failures were reoperated upon, another 16 were not treated by re-resection.
Comment The generally good and satisfactory results of surgical treatment of postintubation stenosis (93.7%), even when it involves the subglottic larynx (90.3%) and even in the presence of the rare TEF justify resection and reconstruction as treatments of choice.32 Good results are confirmed in reports by and Maddaus and colleagues,33 Pearson and Andrews,35 Couraud and colleagues,39 and Bisson and colleagues.40 These excellent series are not further detailed here because they report findings very similar to those described. T tubes, inlying stents, and laser treatment may be applicable in a very limited spectrum of lesions and at a much lesser level of success. Laser treatment most often fails as a definitive treatment. The “thin, web-like stenosis,” in which laser treatment might be expected to effect cure, is an extremely rare lesion (see Chapter 37, “Laser Therapy for Tracheobronchial Lesions”). Failure rate for resection was 3.9%, with the death rate at 2.4%, for all patients. For the more difficult laryngotracheal cases, these rates are 8.1% and 1.6%, respectively, counseling caution in these patients. The lower surgical success rate in patients who had prior failure of reconstruction confirms the observation that the first operation is most likely to succeed and should ideally be performed by experienced hands. The more complex the prior treatment, the more likely eventual failure, even after reoperation. Finally, it must be emphasized that a permanent tracheal T tube may be the best solution for a patient with extensive tracheal damage that defies straightforward reconstruction.
References 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11.
Briggs BD. Prolonged endotracheal intubation. Anesthesiology 1950;11:129–31. Lindholm C-E. Prolonged endotracheal intubation. Acta Anaesth Scand 1969;13 Suppl 33:1–131. Whited RE. A prospective study of laryngotracheal sequelae in long-term intubation. Laryngoscope 1984;94: 367–77. Donnelly WH. Histopathology of endotracheal intubation. An autopsy study of 99 cases. Arch Pathol 1969;88:511–20. Bergström J, Moberg A, Orell SR. On the pathogenesis of laryngeal injuries following prolonged intubation. Acta Otolaryngol 1962;55:342–6. Schmidt VW, Schaap RN, Mortensen JD. Immediate mucosal effects of short term, soft-cuff endotracheal intubation. Arch Pathol Lab Med 1979;103:516–21. Hedden M, Ersoz CJ, Donnelly WH, Safar P. Laryngeal tracheal damage after prolonged use of orotracheal tubes in adults. JAMA 1969;207:703–8. Grillo HC, Mathisen DJ, Wain JC. Laryngotracheal resection and reconstruction for subglottic stenosis. Ann Thorac Surg 1992;53:54–63. Brantigan CO, Grow JB. Cricothyroidotomy: elective use in respiratory problems requiring tracheotomy. J Thorac Cardiovasc Surg 1976;171:72–81. Kuriloff DB, Setzen M, Portnoy W, Gadaleta D. Laryngotracheal injury following cricothyroidotomy. Laryngoscope 1989;99:125–30. Andrews MJ, Pearson FG. Incidence and pathogenesis of
12. 13.
14.
15. 16. 17. 18. 19.
tracheal injury following cuffed tube tracheostomy with assisted ventilation. Analysis of a two year prospective study. Ann Surg 1971;173:249–63. Cooper JD, Grillo HC. The evolution of tracheal injury due to ventilatory assistance through cuffed tubes. A pathologic study. Ann Surg 1969;169:334–8. Florange W, Muller J, Forster E. Morphologie de la nécrose trachéale après trachéotomie et utilisation d’une prothèse respiratoire. Anesth Analg Réan 1965;22:693–703. Grillo HC, Cooper JD, Geffin B, Pontoppidan H. A lowpressure cuff for tracheostomy tubes to minimize tracheal injury: a comparative clinical trial. J Thorac Cardiovasc Surg 1971;62:898–907. Cooper JD, Grillo HC. Experimental production and prevention of injury due to cuffed tracheal tubes. Surg Gyn Obstet 1969;129:1235–41. Grillo HC. The management of tracheal stenosis following assisted respiration. J Thorac Cardiovasc Surg 1969;57:52–71. Stetson JB, Guess WL. Causes of damage to tissues by polymers and elastomers used in the fabrication of tracheal devices. Anesthesiology 1970;33:635–52. Shelly WM, Dawson RC, May IA. Cuffed tubes as a cause of tracheal stenosis. J Thorac Cardiovasc Surg 1969;57:623–7. Murphy DA, MacLean LD, Dobell ARC. Tracheal stenosis as a complication of tracheostomy. Ann Thorac Surg 1966;2:44–51.
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20. 21. 22. 23. 24. 25. 26. 27. 28. 29. 30.
31.
Harley HRS. Laryngotracheal obstruction complicating tracheostomy or endotracheal intubation with assisted respiration. A critical review. Thorax 1971;26:493–533. Tobin MJ. Advances in mechanical ventilation. N Engl J Med 2001;344:1986–96. Adriani J, Phillips M. Use of the endotracheal cuff: some data pro and con. Anesthesiology 1957;18:1–14. Knowlson GTG, Bassett HFM. The pressures exerted on the trachea by endotracheal inflatable cuffs. Br J Anaesth 1970;42:834–7. Carroll R, Hedden M, Safar P. Intratracheal cuffs: performance characteristics. Anesthesiology 1969;31:275–81. Lomholt N. A new tracheostomy tube. I. Cuff with controlled pressure on the tracheal mucous membrane. Acta Anaesth Scand 1967;11:311–8. Geffin B, Pontoppidan H. Reduction of tracheal damage by the prestretching of inflatable cuffs. Anesthesiology 1969;31:462–3. Ching NPH, Nealon TF. Clinical experience with new low-pressure high volume tracheostomy cuffs. N Y State J Med 1974;74:2379–84. Ching NPH, Ayres SM, Paegle RP, et al. The contribution of cuff volume and pressure in tracheostomy tube damage. J Thorac Cardiovasc Surg 1971;62:402–10. Kamen JM, Wilkinson CJ. A new low-pressure cuff for endotracheal tubes. Anesthesiology 1971;34:482–5. Magovern GJ, Shively JG, Fecht D, Theroz F. The clinical and experimental evaluation of a controlled-pressure intratracheal cuff. J Thorac Cardiovasc Surg 1972; 64:747–56. Arens JF, Ochsner JL, Gee G. Volume-limited intermittent
32. 33. 34.
35. 36. 37. 38.
39. 40.
cuff inflation for long-term respiratory assistance. J Thorac Cardiovasc Surg 1969;58:837–41. Grillo HC, Donahue DM, Mathisen DJ, et al. Postintubation tracheal stenosis: treatment and results. J Thorac Cardiovasc Surg 1995;109:486–93. Maddaus MA, Toth JLR, Gullane PJ, et al. Subglottic tracheal resection and synchronous laryngeal reconstruction. J Thorac Cardiovasc Surg 1992;104:1443–50. Gaissert HA, Grillo HC, Wright CD, et al. Lengthening and complication of benign tracheobronchial strictures by self-expanding microinvasive ultraflex and wall stents. J Thorac Cardiovasc Surg 2003. [In press] Pearson FG, Andrews MJ. Detection and management of tracheal stenosis following cuffed tube tracheostomy. Ann Thorac Surg 1971;12:359–74. Dedo HH, Fishman NH. Laryngeal release and sleeve resection for tracheal stenosis. Ann Otol Rhinol Laryngol 1969;78:285–8. Montgomery WW. Suprahyoid release for tracheal stenosis. Arch Otolaryngol 1974;99:255–60. Donahue DM, Grillo HC, Wain JC, et al. Reoperative tracheal resection and reconstruction for unsuccessful repair of post intubation stenosis. J Thorac Cardiovasc Surg 1997;114:934–9. Couraud L, Jongon JB, Velly JF. Surgical treatment of nontumoral stenosis of the upper airway. Ann Thorac Surg 1995;60:250–60. Bisson A, Bonnette P, El Kadi B, et al. Tracheal sleeve resection for iatrogenic stenoses (subglottic laryngeal and tracheal). J Thorac Cardiovasc Surg 1992; 104:882–7.
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C H A P T E R T W E LV E
Acquired Tracheoesophageal and Bronchoesophageal Fistula Hermes C. Grillo, MD
Acquired Tracheoesophageal Fistula Bronchoesophageal Fistulae
Acquired Tracheoesophageal Fistula Granulomatous infection, foreign bodies, and trauma used to be the most common causes of benign acquired tracheoesophageal fistula (TEF). Added to these were fistulae from complications of surgical procedures such as anterior cervical spine fusion and laryngectomy. With widespread use of cuffed tubes for ventilation, postintubation fistulae became predominant. In 1968, in a review of acquired nonmalignant esophagotracheal and esophagobronchial fistulae, Wesselhoeft and Keshishian reported no cases of tracheoesophageal fistula related to cuffs.1 By 1973, Thomas collected 46 such cases (30 fully documented), including 7 of his own.2 Although the use of low-pressure, large-volume cuffs has reduced the incidence, fistulae from this source remain the most common. Immunodeficiency syndromes may also result in fistulae. In fistulae due to granuloma and foreign body, the pathology involves the membranous wall of the trachea and is often limited in extent.3 Traumatic fistulae may be very extensive and be accompanied by mediastinal inflammation and infection. A postintubation fistula results from erosion of the membranous wall of the trachea and the adjacent esophageal wall, “the party wall,” because of pressure from the ventilatory cuff usually exerted against a firm nasogastric tube lying in the esophagus (Figures 12-1, 12-2) (see Chapter 11, “Postintubation Stenosis”). Overinflation of a large-volume cuff by even a small, added volume of air converts it to a high-pressure cuff. The fistula may erode the entire width of the membranous wall; these are often termed “giant fistulae.” Since the inflammatory process is progressive, there is never leakage into the mediastinum in the way there is in a traumatic fistula. Spontaneous healing of such fistulae has not been documented, although on rare occasion, a small recent traumatic fistula may close spontaneously. Circumferential injury to the trachea is almost always present concurrently with a postintubation TEF due to pressure necrosis caused by the cuff. Fistula from the neoesophagus to trachea after esophagectomy is fortunately rare.4 These may follow anastomotic leakage, dilation of stenosis, or tracheal or enteric ischemia as a result of surgical dissection.5 Cervical anastomotic leakage is a principal cause. Symptoms range from cough associated with ingestion to life threatening aspiration pneumonia.
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A
B
FIGURE 12-1 Origin and anatomy of postintubation tracheoesophageal fistula. Lateral diagrams of the trachea and esophagus. A, The overdistended cuff has injured the trachea circumferentially. The “party wall” posteriorly has become devascularized and has necrosed by being pinched between the cuff and a firm nasogastric tube in the esophagus. B, The fistula is usually below the stoma, at the level of the balloon cuff.
More recently, expandable stents have caused TEF spontaneously, after placement of an expandable coated esophageal stent, and as a consequence of difficult removal of a tracheal stent which has become severely obstructive.6,7 The rare but devastating occurrence of necrotizing esophagitis that occurs in immunodeficiency states produces both TEF and bronchoesophageal fistula (BEF), with a high mortality rate.8 Malignant TEF often results from carcinoma of the esophagus, and less so from carcinoma of the lung or lymphoma. Isolated instances have been associated with adenoid cystic carcinoma and even carcinoid tumor. Irradiation treatment of esophageal carcinoma that involves the tracheal wall may accelerate fistula formation. The topic of congenital tracheoesophageal fistula and esophageal atresia has been extensively treated in many textbooks of surgery and pediatric surgery. It is not included in this book, especially since the reconstructive problem is largely esophageal. Rarely, a small recurrent fistula may become symptomatic many years after repair of a congenital TEF in infancy. Attention should be called, however, to the very rare and usually cervical congenital H-type fistula without esophageal atresia, which may give symptoms later in life (see Chapter 6, “Congenital and Acquired Tracheal Lesions in Children”). The signs are principally cough or choking episodes after ingestion of liquid or other food. The fistula may be very small and clini-
Acquired Tracheoesophageal and Bronchoesophageal Fistula
cal presentation not obvious. Repeated bouts of respiratory infection may occur. Suspicion leads to diagnosis by bronchoscopy and radiography.9,10 A description is included at the end of this chapter of the rare entity of bronchoesophageal fistula, both congenital and acquired.
Clinical Presentation and Diagnosis Benign Fistula. If a fistula develops in a patient on a respirator, a sudden increase in secretions is often noted as saliva enters the airway. It becomes difficult to maintain a seal with the cuff. Pulmonary infiltrates and pneumonia follow. Respiratory insufficiency may worsen. Cough follows swallowing. With ventilation, air may be heard escaping into the pharynx and the abdomen may become distended. Gastric feedings may appear on tracheal suctioning. Gastric reflux into the lungs can be disastrous and eventually fatal. If the patient is receiving oral feedings, these will appear in the tracheal suctioning. Chest x-ray commonly shows the esophagus to be dilated distal to the fistula and the stomach may be dilated (Figure 12-3). A swallow of water stained with methylene blue will appear in the tracheostomy. This test is to be interpreted with caution since aspiration of dye into the larynx produces the same result. Fluoroscopy by an experienced radiologist, with ingestion of a small amount of barium, usually delineates the level and approximate size of the fistula (Figures 12-4, 12-5). The fistula may be visible directly through a tracheostomy if it is present. Bronchoscopy should be done promptly if a fistula is suspected. In a patient who is on a respirator, a flexible bronchoscopy may be performed through an endotracheal tube, which is withdrawn just sufficiently to allow visualization of the
FIGURE 12-2 Postmortem specimen of the trachea of a patient on long-term ventilation through a tracheostomy tube with a highpressure cuff. A large membranous wall fistula has formed (arrow). The tracheal stoma is superior. Note the circumferential cuff damage.
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FIGURE 12-3 Roentgenogram of a patient with postintubation tracheoesophageal fistula. A, Note the distended esophagus, typical of this condition. Fistula is visible as a radiolucent circle. B, After repair, a normal tracheal air column is seen. Arrows mark the glottic level in both roentgenograms.
fistula. The same may be done through a tracheostomy tube while continuing ventilation in both cases. Passage of a rigid ventilating bronchoscope via the larynx allows the best assessment of the entire airway and locates the fistula relative to the cricoid and carina. The lengths of the fistula and of the normal airway are measured. A postintubation fistula is usually clearly identifiable (Figure 12-6). If not, methylene blue in saline may be instilled into the upper esophagus with the caution already noted to avoid overfilling and aspiration. Esophagoscopy is less likely to offer a good view, especially of smaller fistulae. A postintubation fistula usually lies a centimeter or two below the level of a tracheal stoma, since the fistula is located at the cuff site (see Figure 12-1). Chronic fistulae from other causes present with cough on fluid or food ingestion, pulmonary infection, and occasional hemoptysis. Contrast images and bronchoscopy are diagnostic but most important is the clinician’s suspicion of a fistula. This is even more critical after severe chest trauma since a tracheoesophageal rupture may go unrecognized, or a fistula may be delayed in its formation, until life threatening mediastinal sepsis is established. Malignant Fistula. In an excellent review of 207 malignant esophagorespiratory fistulae, Burt and colleagues confirmed esophageal carcinoma as the primary neoplasm (78%).11 The tumors were located principally in the upper thoracic esophagus and were principally squamous in histology. The incidence of fistulization in this series of esophageal carcinomas was 4.5%. Lung cancer accounted for 16% of the malignant fistulae, and in only 3 patients were tracheoesophageal fistulae related to primary tracheal neoplasms. Other neoplasms that were associated with a tracheoesophageal fistula were Hodgkin’s disease, metastatic breast cancer, and laryngeal carcinoma.12,13 All patients with carcinoma of the upper- or midesophagus should undergo bronchoscopy in their initial work-up. If an abnormality is identified between the trachea and esophagus radiographically, on
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345
A
B FIGURE 12-4 Postintubation laryngotracheal stenosis with tracheoesophageal fistula (TEF) in a 56-year-old man. He had suffered cricoidostomy and failed TEF repair prior to referral. A, Tomograms of stenosis. Anteroposterior view on the left shows a deformed larynx with maximum stenosis at the laryngotracheal junction. The lateral view on the right demonstrates a narrowed subglottic larynx, with the most severe stenosis just below this (arrow) and above the stomal tract. B, Barium swallow in the same patient. The preoperative view on the left shows the fistula (arrow). On the right is a postoperative view of the repaired esophagus. Laryngotracheal resection of the stenosis and reconstruction were performed at the same time as closure of the fistula. There is no aspiration. Strap muscle was interposed between the two suture lines.
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computed tomography (CT) scan or, most definitively, by ultrasonography, an abnormality is likely to be seen bronchoscopically; that is, compression, induration, granularity, or infiltration. If tumor is identified or suspected, the possibility of a fistula may be anticipated. Cough, hemoptysis, fever, and aspiration all signal malignant fistulization. Bronchoscopy with the use of methylene blue and contrast esophagography will show the communication. Esophagoscopy is often less definitive because of the bulk of tumor adjacent to the fistula. The fistula may be tracheal (53%) or bronchial (38%), and a few are pulmonary (6%).11 Frequently, the fistula follows prior radio- or chemotherapy, as might be expected, since treatment destroys tumor which had previously destroyed normal tissue. Although the disease may well be localized rather than disseminated at the time of manifestation of a fistula, progression of aspiration, pneumonia, lung abscess, and asphyxiation can be rapid. The clinical course typically is measured in weeks and months.
Management It hardly needs to be said that major efforts must be made in all of these patients to clear up local and pulmonary sepses and to improve nutrition prior to surgical procedures. Benign fistulae not related to ventilation are individually managed depending on their cause, size, location, and degree of surrounding pathology. Cervical (with the possibility of partial upper sternal division) or, less frequently, right transthoracic approaches are used, depending upon the level of the fistula (Figure 12-7). Only a supracarinal fistula requires thoracotomy. Principles of closure of the TEF include complete dissection of the fistula, its division, effectively planned
A
B
FIGURE 12-5 Small chronic upper tracheoesophageal fistula in a 66-year-old woman resulting from foreign body ingestion at age 8, which when she was age 25 was endoscopically removed by Dr. Chevalier Jackson. A, The upper arrow in the contrast esophagogram indicates the cricopharyngeus, and the lower arrow indicates the fistula, in anterior view. B, Lateral view; fistula is indicated by the arrow.
Acquired Tracheoesophageal and Bronchoesophageal Fistula
FIGURE 12-6 Bronchoscopic view of postintubation tracheoesophageal fistula. Note the large size of the membranous wall defect and also that there is circumferential tracheal damage at the level of the fistula. Normal tracheal rings are visible distally. One-stage repair was done with esophageal closure and tracheal resection and reconstruction. Also, see Figures 31 and 32 (Color Plate 15).
membranous wall suture closure which is tension-free, and two-layered esophageal closure (see Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula”).14 Recurrent fistulization is avoided by interposition of healthy pedicled tissue (such as a strap muscle in the neck or intercostal muscle in the chest) between the tracheal and esophageal suture lines. The technique of borrowing adjacent esophageal wall to facilitate closure without tension is also discussed in Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula.” Recurrent laryngeal nerves must be carefully avoided. If the fistula is of any extent, the trachea may be best and most safely managed by resection of the segment containing the fistula, with endto-end tracheal anastomosis after esophageal closure, even if circumferential tracheal damage is not present. Post-traumatic fistulae may be extensive, accompanied by mediastinal injury and infection. Decision on therapy in these injuries must be individualized (see Chapter 9, “Tracheal and Bronchial Trauma”). In the most severe and delayed post-traumatic cases, esophageal exclusion may have to be considered, an alternative that is usually unnecessary in other types of TEF.14 Surgical technique is described in Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula.” Postsurgical fistulae following esophagectomy are treated with respect to the location and size of the fistula, the presence or absence of necrosis in the neoesophagus, mediastinitis, and the severity of symptoms. Treatment may range from drainage with conservative management, local tissue excision with buttressed closures, to removal of the neoesophagus, and reconstruction of a new esophageal replacement, possibly in stages.4 Esophagorespiratory fistula due to necrotizing esophagitis, from infection in immunocompromised patients, requires esophagectomy.8 An attempt to close a postintubation fistula in a patient who is still on a respirator is almost certain to fail. Prolonged ventilation after tracheal reconstruction is likely to encourage dehiscence or stenosis. These patients are best managed conservatively, with every effort made to wean them from mechanical ventilation to permit later definitive surgical repair. If an esophageal tube is present, it is withdrawn. If possible, the tracheostomy cuff is situated just below the fistula, using as little pressure as possible to obtain a seal. A draining gastrostomy is positioned to avoid aspiration of gastric contents and a jejunostomy is placed for feeding. The head of the bed is kept in an elevated position. Vigorous efforts are made to clear any pulmonary infection. Under this regimen, the situation usually improves quite rapidly. The small amount of saliva that continues to trickle into the respiratory tree seems to be handled comparatively well with the help of frequent
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B
FIGURE 12-7
Chronic fistula due to foreign body ingestion in childhood shown in Figure 12-5. Endoscopic findings and treatment. A, Bronchoscopic visualization of the small fistula (arrow) demonstrated in Figure 12-5, due to foreign body erosion. Compare this with the postintubation tracheoesophageal fistula in Figure 12-6. B, Esophagoscopic view of the same fistula (arrow). The small lesion is difficult to see in the esophageal folds. C, Right cervical operative exposure. The diagram clarifies the anatomy. A Penrose drain passes beneath the fistulous tract, which emerges from the posterolateral wall of the trachea on the right. Sutures have been placed in the tract closer to the esophagus (on the left) to leave more tissue for closure of the tracheal wall. The vascular loop passes beneath a nonrecurrent right inferior laryngeal nerve. This was critical since the left nerve had been injured in a prior failed attempt at another hospital to close the fistula from the left side. The right lobe of thyroid is retracted with a heavy suture at the left. Pedicled sternohyoid muscle was interposed between the tracheal and esophageal suture lines.
tracheal suctioning. Esophageal diversion is almost never necessary. If it is required under highly unusual circ*mstances and is feasible, a disconnecting procedure is preferred to in-continuity esophagostomy. The proximal end of the esophagus is brought out laterally (left neck) as a salivary fistula and the distal end turned in with care. Since most of these fistulae are high in location, the point of division should be immediately above the fistula to simplify later reconstruction by leaving sufficient proximal esophagus. More often
Acquired Tracheoesophageal and Bronchoesophageal Fistula
than not, however, the fistula is located so close to the cricopharyngeus that exteriorized esophagostomy is impossible. The lower end of the esophagus at the gastric inlet should not be ligated, stapled, or divided. Continuous suction on the gastrostomy is usually sufficient to protect the trachea from reflux of gastric juices. The gastrostomy also serves to keep the stomach from becoming distended. After weaning, surgical correction includes closure of the esophageal fistula in layers, resection of the circumferentially damaged tracheal segment and its reconstruction, plus interposition of viable tissue between the two suture lines. This is all performed in a single stage (Figure 12-8).15,16 Even though the transverse tracheal anastomotic suture line and the vertical esophageal suture line may be at different levels, it always seems safer to use an interposition flap, as described. I have seen no difficulties arising from these flaps. The precise technique of repair and methods of dealing with special technical problems are detailed in Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula.” In rare cases where the tracheal injury is too long to permit tracheal reanastomosis, the esophagus is closed nonetheless to eliminate the fistula and tracheal patency, and function is restored with a permanent T tube. If laryngotracheal stenosis is present, that is managed in the usual way for such lesions after closure of the esophagus (see Figure 12-4) (see Chapter 25, “Laryngotracheal Reconstruction”). In the past, it was recommended by some authors that the esophageal aperture be closed in an initial operation, and that any tracheal process be dealt with at a second procedure.2 There is no justification for this approach.14–16 Not only does the tracheal lesion require resection in any case, but both the proposed initial and later operations become much more difficult if staged. Malignant fistula is most often best treated by palliative bypass intubation, given the patient’s limited expectation for life. Exclusion of a segment of fistulized esophagus, with concomitant intestinal bypass of the esophagus, is only very rarely advisable either in a patient in very good condition or in
FIGURE 12-8
Operative repair of postintubation tracheoesophageal fistula (TEF). At this stage, the circumferentially damaged segment of trachea wherein the TEF was located has been resected. An Allis forceps elevates the proximal end of trachea. The esophagus, held in forceps, is ready for meticulous closure. Sutures identify the proximal and distal margins of the defect. Tracheal anastomosis will then be done, after pedicled sternohyoid muscle interposition over the esophageal suture line. (See Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula,” for operative details.)
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one with a rare slow-moving tumor of unusual type. An intermediate alternative of intubation with partial exclusion by esophagostomy and with gastrostomy plus jejunostomy (for prevention of aspiration and for nutrition) may be considered, with the possibility of a later restitution of gastrointestinal continuity by substernal gastric or colonic transposition. Duranceau and Jamieson carefully reviewed these options in 1984, and Burt and colleagues described their experiences in 1991.11,17 In a terminal and debilitated patient, abstinence from any intervention may be the kindest therapeutic choice. If intubation through the tumor is elected, pulsion rather than traction (via a gastrostomy) seems to be the best palliative maneuver. Intubation techniques are not without morbidity and mortality. Tubes must be of impervious material or be coated if of an expandable type, in order to prevent prompt ingrowth of tumor through interstices. In 1920, Kirschner used the stomach anastomosed to the cervical esophagus to bypass a malignant fistula.18 Many variations have since been employed, using the stomach, jejunum, and colon for interposition as well as extracorporeal synthetic tubes. An esophagus excluded above the fistula, and below by occlusion of the gastroesophageal junction, has too often produced copious secretions, leaked, or ruptured. A residual esophagus should therefore be drained with a loop or arm of jejunum if left in situ. A preferable technique is to divide the esophagus just below the fistula as well as above, creating a smaller “diverticulum” at the fistulous site, and excise the distal esophagus (via a transhiatal approach). The stomach and colon are the favored gastrointestinal replacement conduits, and are placed substernally (see Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula”).11,17,19 Each patient must be carefully evaluated and treatment individualized, bearing in mind that any treatment will at best be palliative and usually for a short term. If radical treatment is to be offered, it should be instituted promptly.
Results Benign Acquired Fistula. The difficult problem of treatment of a benign acquired tracheoesophageal fistula, and the particularly threatening one of fistula due to intubation and ventilation, may be successfully managed for the most part by attention to the principles stated and using the techniques detailed in Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula.” The relatively few reports of any number of patients treated in accordance with these principles, which were enunciated by Grillo and colleagues in 1976, are all encouraging.15 Couraud and colleagues, Dartevelle and Macchiarini, and Mathisen and colleagues, in a total of 78 patients, summarized by Dartevelle and Macchiarini, performed simple closure of fistula in 29, closure with tracheal resection in 44, and diversion in only 5 patients.14,20,21 Recurrences of TEF were at the rate of 6.4 to 8.3% and mortality at 6.3 to 12.5% (Table 12-1). Macchiarini and colleagues commented on the superiority in their experience of
Table 12-1 Results of Operative Repair of Postintubation Tracheoesophageal Fistulae (TEF) Type of Operation
Author
Number of Patients
Simple Closure
TR + EC
ED
TEF Recurrence (%)
Mortality (%)
Mathisen et al14 Couraud et al20 Dartevelle and Macchiarini21
38 16 24
9 9 11
29 5 10
0 2 3
3 (7.9) 0 2 (8.3)
4 (10.5) 1 (6.3) 3 (12.5)
Total
78
29
44
5
5 (6.4)
12 (10.3)
Data from Dartevelle P and Macchiarini P.21 EC = esophageal closure; ED = esophageal diversion; TR = tracheal resection.
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the anterior approach as well as the definitive single-stage repair by our technique over other types of surgical repairs of varying complexity.15,22 In our series of 38 patients, 27 TEFs resulted from ventilation, 5 from laryngotracheal trauma, 2 from anterior spine fusions, and 2 from irradiation after prior laryngectomy for cancer.14 Foreign body and possible congenital origin accounted for 2 more. Eight had one or more prior failed repairs, 7 had esophageal diversions, 3 had occlusion of the esophagogastric junction, and 1 had colon interposition. Approaches were by collar incision in 26, partial sternotomy in 10, and complete sternotomy or lateral thoracotomy in a few patients with special problems. Tracheal resection was done in 31 patients and laryngotracheal resection and reconstruction in 5. Laryngeal release was necessary in only 2 patients. Where there was insufficient tracheal mucosa for tracheal closure, adjacent esophageal mucosa was borrowed (see Chapter 26, “Repair of Acquired Tracheoesophageal and Bronchoesophageal Fistula”). In a patient where there was insufficient length of trachea for reconstruction, the TEF was closed, the repair buttressed, and the airway reestablished with a permanent T tube. Prior cervical esophagostomy necessitated end-to-end esophageal anastomosis in 5 patients. Three deaths followed transthoracic repair of distal fistulae in the face of mediastinal sepsis (due to trauma), and 1 patient died from dehiscence after an extended tracheal resection. Two patients had successful reoperation for recurrent TEF and one healed a further small recurrence spontaneously. Two patients needed temporary tracheostomy. Postoperative aspiration in some patients gradually resolved. Thirty-three of 34 operative survivors were successful. Three of 4 with vocal cord problems had these conditions prior to operation. These outcomes of success in patients, many made more complex by prior failed surgery, indicate that generally successful results can be obtained in this difficult group of patients. Clearly, esophageal diversion is almost never necessary and, equally clearly, a single-staged procedure is indicated where tracheal injury accompanies the TEF. Closure of a fistula should be accomplished after a patient has been weaned from the respirator. Malignant Fistula. In their review, Duranceau and Jamieson cited complication and mortality rates in the order of 28 to 38% and 6 to 17%, respectively, for “push through” palliative intubation.17 Control of aspiration pneumonia is not universal and does not last as the disease progresses. Early death still occurs in many patients, although with the best results, symptoms are controlled for a time and oral ingestion becomes possible. There is no clear-cut advantage to the older “pull through” method; it has the disadvantages of another procedure and probably more complications. Burt and colleagues found a median survival of 5 weeks (35 days) in 207 patients receiving all modes of treatment.11 The primary neoplasm and location of the fistula were not determinants of survival. Specific treatment extended the median survival from 22 to 47 days. Endoprosthesis did not alter the median survival. Supportive care included all minor methods but not functional exclusion of the fistula. Chemotherapy and radiotherapy alone led to slightly longer survival, probably by eliminating the negative effects of procedures. Esophageal exclusion alone was no better, but gastric or colonic bypass, done in a small number of patients, increased survival to a 77-day median, a significant but not overwhelming improvement in exchange for very extensive surgery. The overall 30-day mortality was 46%. Pulmonary sepsis was the principal cause of death in over 80%, with bleeding in 12%.
Bronchoesophageal Fistulae Bronchoesophageal fistulae (BEF) are rare lesions, either congenital or acquired. The congenital fistulae can be insidious and may go unrecognized for years. They are more frequently recognized in the adult than in the child. Diagnosis of both congenital and acquired lesions depends upon a high index of suspicion.
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Congenital Bronchoesophageal Fistula Braimbridge and Keith classified BEF into four types (Figure 12-9).23 Type I seems to result, at least sometimes, from inflammatory changes in an esophageal diverticulum, which then secondarily fistulizes to a bronchus. Although some diverticula are undoubtedly congenital, traction diverticula are also likely represented in this group of lesions and the fistula itself may not therefore always be of congenital origin. Nonetheless, I retain their classification of “congenital” BEF’s. Type I presents with a small fistula, at times inflamed, at the tip of an esophageal diverticulum. Type II is a simple fistula extending from the esophagus at an upward angle to a bronchus. This is by far the most common type. Next in incidence is type III, where the fistula connects to a cyst in the lung and thence to bronchi. Type IV is a fistula to a sequestrated lung, which is supplied by a systemic artery. The tract runs most commonly from the middle third of the esophagus, and less often from the lower third to the right lower lobe (either segmental or lobar bronchus), while half as often to the bronchi of the left lower lobe and in diminishing frequency to the bronchus intermedius, left main bronchus, right middle lobe, and right upper lobe bronchi. In a review of 100 cases reported up to 1990, Risher and colleagues found only 5 cases of type III and 3 of type IV.24 Congenital fistulae are characterized by lack of inflammation, absence of adherent or inflamed lymph nodes, and a tract lined with squamous or columnar mucosa with adjacent muscularis mucosa. Clinical Presentation and Diagnosis. Distribution of patients is about equal for males and females, with 75% of patients over 17 years of age. Paroxysmal cough, cough on ingestion of food especially after liquids, frequent respiratory infections, hemoptysis, and hematemesis are found.25–27 Symptoms may be largely of cough and respiratory infection for many years, leading to a delay in diagnosis from 5 to 30 years. Retrospectively, a long history is usually identified. Delay in appearance of severe symptoms has been attributed to the oblique course of the tract and to its possible initial obstruction either by a thin membrane, which later ruptures after inflammation, or to a mucosal flap valve in the tract. Marked delay in the discovery of a fistula can lead to death from recurrent pulmonary suppuration. Treatment is therefore urged as soon as diagnosis is made. Diagnosis is made by contrast esophagography (Figure 12-10) and bronchoscopy, sometimes with methylene blue instillation into the esophagus. Esophagoscopy is less often definitive. Insufflation of gases into the trachea during esophagoscopy may aid in pinpointing a tiny fistula. Contrast should be introduced for esophagography with the patient in a position where cough ordinarily follows oral ingestion, rather than with the patient in a recumbent position. CT scan helps to assess pulmonary damage.
12-9 Braimbridge and Keith’s classification of congenital bronchoesophageal fistula: type I, wide neck diverticulum with inflammatory fistula at tip; type II, simple fistula, the most common type; type III, fistula with cyst; type IV, fistula with sequestration of lung.23
FIGURE
Acquired Tracheoesophageal and Bronchoesophageal Fistula
353
Treatment. Despite a few reports of alternative treatments, surgical excision of the tract and closure of the fistula at either end is the best choice (Figure 12-11). Right or left thoracotomy is selected according to the location of the fistula. Two layers of 4-0 Vicryl sutures are advised for closure of the esophageal end, or one layer of staples with a sutured second layer. One layer of Vicryl is used on the bronchial side. A substantial flap of healthy tissue, such as a pericardial fat pad or intercostal muscle, is pedicled between the two suture lines. Simple stapling without division of the tract is likely to result in recurrent fistula. Any irremediably
FIGURE 12-10 Contrast esophagogram demonstrating congenital bronchoesophageal fistula (arrow) in a 65-year-old woman. The fistula opened into the left lower lobe bronchus just medial to the superior segmental bronchus.
FIGURE 12-11 Operative repair of type I congenital bronchoesophageal fistula via left thoracotomy. A Penrose drain encircles the dissected fistula and esophageal diverticulum. The esophagus is at the left, the lung at the right. The diverticulum was excised, both sides of the channel sutured, and intercostal muscle pedicled between.
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injured portion of lung is resected at the same time. This has been necessary in many cases. Results of treatment are generally excellent.
Acquired Bronchoesophageal Fistula Bronchoesophageal fistula can result from trauma or infection or rare additional causes.1,28–30 Blunt chest trauma has produced a BEF, although the cervical or supracarinal trachea is more likely to be a locus of posttraumatic esophagorespiratory fistula than a bronchus. Instrumentation (such as variceal sclerosis), chemical burns (lye ingestion), and foreign bodies have all been implicated. Infectious agents associated with fistula include tuberculosis, histoplasmosis (Figure 12-12), actinomycosis, and syphilis. Often, these are associated with inflammatory lymph nodes or broncholithiasis. Nontuberculous empyema, suppurative esophagitis, and infected bronchogenic cysts are other etiologies. Fistula has also been associated with traction diverticula related to lymph nodes. Fistulae have occurred due to necrotizing vasculitis and silicotic nodules.1,28–30 Clinical presentation, diagnosis, and treatment are much the same as described for congenital BEF. Results of surgical treatment are very satisfactory (Figure 12-13).28–30 Since more inflammation may be encountered, and more dissection may be necessary to remove involved lymph nodes than in uninflamed congenital fistulae, I favor interposition of an intercostal muscle flap in these cases. The flap is raised at the time of initial thoracotomy. An associated esophageal diverticulum is resected and the esophagus repaired in two layers. Concomitant pulmonary resection is dictated by irretrievable lung damage from chronic infection. In our series of 9 patients collected over 41 years, 4 followed thoracic surgery, 3 were due to histoplasmosis, and 1 each were due to silicosis, foreign body, lye ingestion, bronchogenic cyst, and esophageal diverticulum, respectively.30 One BEF was congenital. The patient with lye ingestion succumbed. There were no recurrences after successful surgical closures, performed as described above. During this same period of time, 215 patients were recorded with BEF due to bronchogenic or esophageal malignancy.
12-12 Bronchoesophageal fistula due to histoplasmosis. (Courtesy of Dr. Delos M. Cosgrove III.)
FIGURE
Acquired Tracheoesophageal and Bronchoesophageal Fistula
FIGURE 12-13 Acquired bronchoesophageal fistula due to broncholithiasis from silicosis involving mediastinal lymph nodes. A, Barium swallow demonstrates a fistula (arrow) from the esophagus to the bronchus intermedius. B, Computed tomography section shows irregular thickening of the esophageal wall (arrow) and a calcified lymph node protruding into the bronchus intermedius (arrow).
A
B
References 1. 2. 3. 4.
Wesselhoeft CW Jr, Keshishian JM. Acquired nonmalignant esophagotracheal and esophagobronchial fistula. Ann Thorac Surg 1968;6:187–95. Thomas AN. The diagnosis and treatment of tracheoesophageal fistula caused by cuffed tracheal tubes. J Thorac Cardiovasc Surg 1973;65:612–9. Macchiarini P, Delamore N, Beuzeboc P, et al. Tracheoesophageal fistula caused by mycobacterial tuberculosis adenopathy. Ann Thorac Surg 1993;55:1561–3. Buskens CJ, Hulscher JBF, Fockens P, et al. Benign tracheo-neo-esophageal fistulas after subtotal esophagectomy. Ann Thorac Surg 2001;72:221–4.
5. 6. 7.
8.
Fujita H, Kawahara H, Hidaka M, et al. An experimental study on viability of the devascularized trachea. Jpn J Surg 1988;18:77–83. Schawengerdt CG. Tracheoesopahgeal fistula caused by a self-expanding esophageal stent. Ann Thorac Surg 1999; 67:830–1. Gaissert HA, Grillo HC, Wright CD, et al. Lengthening and complication of benign tracheobronchial strictures by self-expanding microinvasive ultraflex and wall stents. J Thorac Cardiovasc Surg 2003. [In press] Gaissert HA, Roper CL, Patterson GA, Grillo HC. Infectious necrotizing esophagitis: outcome after medical
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9. 10. 11. 12. 13.
14. 15. 16. 17. 18. 19.
and surgical intervention. Ann Thorac Surg 2003; 75:342–7. Acosta JL, Battersby JS. Congenital tracheoesophageal fistula in the adult. Ann Thorac Surg 1974;17:51–7. Olivet RT, Payne WS. Congenital H-type tracheoesophageal fistula complicated by achalasia in an adult. Mayo Clin Proc 1975;50:464–8. Burt M, Diehl W, Martini N, et al. Malignant esophagorespiratory fistula: management options and survival. Ann Thorac Surg 1991;52:1222–9. Greven KM, Evans LS. The occurrence and management of esophageal fistulas resulting from Hodgkin’s disease. Cancer 1992;69:1031–3. Tse DG, Summers A, Sanger JR, Haasler GB. Surgical treatment of tracheomediastinal fistula from recurrent Hodgkin’s lymphoma. Ann Thorac Surg 1999; 67:832–4. Mathisen DJ, Grillo HC, Wain JC, Hilgenberg AD. Management of acquired nonmalignant tracheoesophageal fistula. Ann Thorac Surg 1991;52;759–63. Grillo HC, Moncure AC, McEnany MT. Repair of inflammatory tracheoesophageal fistula. Ann Thorac Surg 1976;22:113–9. Hilgenberg AD, Grillo HC. Acquired non-malignant tracheoesophageal fistula. J Thorac Cardiovasc Surg 1983;85:492–8. Duranceau A, Jamieson GG. Malignant tracheoesophageal fistula. Ann Thorac Surg 1984;37:346–54. Kirschner MB. Ein neues Verfohren der Oesophagoplastie. Arch Klin Chir 1920;114:606. Symbas PN, McKeown PP, Hatcher CR Jr, Vlasis SE. Tracheoesophageal fistula from carcinoma of the esophagus. Ann Thorac Surg 1984;38:382–6.
20. 21. 22.
23. 24. 25.
26. 27. 28. 29. 30.
Couraud L, Bercovici D, Zanotti L, et al. Traitement des fistules oesophago-trachéales de la réanimation. Ann Chir 1989;43:677–81. Dartevelle P, Macchiarini P. Management of acquired tracheoesophageal fistula. Chest Surg Clin North Am 1996;6:819–36. Macchiarini P, Verhoye JP, Chepelier A, et al. Evaluation and outcome of different surgical techniques for post-intubation tracheoesophageal fistulas. J Thorac Cardiovasc Surg 2000;119:268–76. Braimbridge MV, Keith HZ. Oesophago-bronchial fistula in the adult. Thorax 1965;20:226–33. Risher WH,Arensman RM, Ochsner JL. Congenital bronchoesophageal fistula. Ann Thorac Surg 1990;49:500–5. Azoulay D, Regnard JF, Magdeleinat P, et al. Congenital respiratory esophageal fistula in the adult. Report of nine cases and review of the literature. J Thorac Cardiovasc Surg 1992;104:381–4. Rämö OJ, Salo JA, Mattila SP. Congenital bronchoesophageal fistula in the adult. Ann Thorac Surg 1995;59:887–90. Kim JH, Park K, Sung SW, Rho JR. Congenital bronchoesophageal fistula in adult patients. Ann Thorac Surg 1995;60:151–5. Wychulis AR, Ellis FH Jr, Andersen HA. Acquired nonmalignant esophagotracheobronchial fistula. JAMA 1966;196:117–22. Spalding AR, Burnery DP, Richie RE. Acquired benign bronchoesophageal fistulas in the adult. Ann Thorac Surg 1979;28:378–83. Mangi AA, Gaissert HA, Wright CD, et al. Benign bronchoesophageal fistula in the adult. Ann Thorac Surg 2002;73:911–5.
C H A P T E R T H I RT E E N
Tracheal Fistula to Brachiocephalic Artery Hermes C. Grillo, MD
Post-Tracheostomy Fistula Postoperative Fistula Diagnosis and Management Results Prevention of a Tracheal-Arterial Fistula
A fistula between the trachea and brachiocephalic (innominate) artery is a rare complication either of tracheostomy, most often in conjunction with ventilatory support,1,2 or of tracheal reconstructive surgery,3,4 or exenteration. External trauma can rupture the artery, produce a false aneurysm or, less often, result in fistula to a lacerated trachea.
Post-Tracheostomy Fistula Two types of tracheoarterial fistulae occur from tracheostomy. The first is due to erosion of the artery lying immediately beneath the curve of the tracheostomy tube. The second is caused by erosion of the anterior tracheal wall into the artery by either the cuff or the tip of the tracheostomy tube. The two lesions must be kept clearly in mind, since the emergency and definitive management of each is different (see Figures 27-1 through 27-3 in Chapter 27, “Repair of Tracheobrachiocephalic Artery Fistula”). Fortunately, the incidence of both of these potentially disastrous lesions has diminished, the first by avoiding errors in the technique of tracheostomy, and the second because of development of large-volume, lowpressure cuffs and their correct usage.
Erosion by a Tracheostomy Tube In children and younger adults, hyperextension of the neck delivers half or more of the trachea into the neck, and the brachiocephalic artery also rises into the base of the neck. If tracheostomy is made with reference to the sternal notch, as was taught in the past, the stoma will be in the midtrachea and will lie just above the artery. The combined thrust of the respirator on the tube, failure to suspend the tube, and arterial pulsation can rapidly lead to arterial erosion by the elbow of the tube. The point of hemorrhage will be at the inferior margin of the stoma. In an emergency, digital pressure is applied at this point to control hemorrhage. Pressure is directed downward and forward against the sternum (see Chapter 27, “Repair of Tracheobrachiocephalic Artery Fistula”). The fistula may occur surprisingly soon after tracheostomy has been done, sometimes in days, although more often in weeks.
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This catastrophe is prevented by correctly locating the tracheostomy at the level of the second and third tracheal rings, using the almost-always palpable cricoid as a landmark. It makes no sense to use the fixed point of the sternal notch with respect to a movable structure, the trachea. In a much less common situation, the uppermost trachea may be nearly contiguous with the brachiocephalic artery in an aged patient with marked kyphosis and very limited cervical extension.
Erosion by a Tracheostomy Tube Cuff or Tip If a tracheostomy is correctly located at the level of the second and third rings, the ventilating cuff or tube tip very often lies behind the point where the brachiocephalic artery crosses the trachea, at about the ninth tracheal ring. If high-pressure cuffs are used, circumferential erosion is sometimes accompanied by sufficient anterior tracheal damage to produce a tracheoarterial fistula. Most often, these lesions follow prolonged ventilation. Less often, a tracheostomy tube that is angulated forward eccentrically, probably due to the distorted way in which a high-pressure cuff might expand, erodes the tracheal wall at this critical point (Figure 13-1). A 90˚ tracheostomy tube is more likely to be thus angulated. Although we continue to see concentric cuff injury by largevolume cuffs used in a high-pressure range (by overinflation), extreme anterior damage seems to occur only rarely nowadays. In these patients, hemorrhage occurs directly into the trachea at a point not accessible to the finger. Emergency control must be obtained by tamponade, by overinflating the tracheostomy tube cuff. An endotracheal tube with an overinflated cuff is more satisfactory since it can be positioned more easily. Management of a post-tracheostomy arterial fistula and of the trachea is detailed in Chapter 27, “Repair of Tracheobrachiocephalic Artery Fistula.” Large-volume, low-pressure cuffs prevent most such injuries nowadays.
Postoperative Fistula Early in the development of tracheal reconstruction, postoperative hemorrhage from the brachiocephalic artery occurred too frequently.3,4 Since cuff stenosis often lies at the level of the artery, the vessel was frequently dissected free from scar and the tracheal anastomosis made immediately behind it. Local infection or erosion at this point of confluence could lead to bleeding. Suture material, a foreign body, probably contributes. One surgeon, who formerly used fine wire for an anterior tracheal anastomosis, attributed some fistulas to abrasion by this unyielding material. Anastomotic dehiscence after tracheal reconstruction, most often managed with subsequent intubation, may expose the artery in what is now an infected space. If tracheal dissection is kept scrupulously close to the trachea and the artery is left undissected with its local tissue investment intact, hemorrhage will almost never follow. When the artery must be dissected because of adherence to tracheal scar, prior tracheal surgery, or in surgery for a neoplasm, it is advisable to place viable tissue, such as an inferiorly based pedicle of sternohyoid muscle or thymus, between the tracheal anastomosis and the overlying artery. The rarity of this complication is seen in its low incidence, occurring in only five (1%) of 503 patients who had tracheal resection and reconstruction for postintubation lesions.5 These instances occurred early in our experience. Late brachiocephalic hemorrhage has long been the bane of mediastinal tracheostomy and exenteration. The artery would become exposed by failure of healing of the skin beneath the mediastinal tracheostomy, sometimes abetted by the effects of prior irradiation. This has largely been prevented by omental coverage or, where indicated, by prophylactic division of the brachiocephalic artery, after preoperative imaging and with intraoperative electroencephalographic monitoring (see Chapter 34, “Cervicomediastinal Exenteration and Mediastinal Tracheostomy”).6 Innominate arteries or even aortic fistulae have resulted over the years from attempts to use tracheal prostheses made of various foreign materials.3 Fixation points of Gianturco tracheobronchial stents have also produced arterial fistulae. Both demonstrate the all too obvious surgical principle of avoiding prolonged pressure on vascular structures by foreign material.
Tracheal Fistula to Brachiocephalic Artery
A 13-1 Tracheobrachiocephalic artery fistula due to anterior erosion by tracheostomy tube cuff. A, Operative field after division and resection of a fistulous arterial segment. The forceps elevates the proximal brachiocephalic arterial stump. The distal artery is visible to the right of the trachea, which exhibits a sizeable anterior defect. The endotracheal tube is visible in the defect. The brachiocephalic vein is retracted caudad with a Penrose drain.
FIGURE
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B 13-1 (CONTNUED) B, The resected segment of circumferentially damaged trachea is at the left. The anterior fistula is visible. The perforated section of the brachiocephalic artery is at the right. Both arterial ends were sutured closed and protected with thymus. A standard tracheal anastomosis was made.
FIGURE
Diagnosis and Management Bleeding immediately after a tracheostomy may result from incomplete hemostasis or from surgically injured branches of cervical vessels. Fresh bleeding thereafter, in any amount, from a tracheostomy or from a postoperative trachea should at once raise the question of innominate artery leakage. Bleeding from the innominate is often massive at the outset. Premonitory minor or limited bleeding may occur however. Only suspicion of the worst will lead to the prompt action necessary. Most instances of bleeding from a tracheostomy are due to granulation tissue, superficial ulceration, or tracheitis. This, however, must be established promptly by bronchoscopy. If bleeding ceases with overinflation of the cuff, a brachiocephalic artery fistula is most likely present. If the hemorrhage is of any moment, the examination is best done in the operating room, with personnel and equipment at hand for instant major intervention. Rigid bronchoscopy is advised, with the tracheostomy tube removed. If blood clot and anterior necrosis is observed, then operation is indicated even if bleeding has ceased at that moment. Examination by a flexible instrument through the tracheostomy tube is unlikely to be satisfactory. Angiography can be of value in showing false aneurysm or rupture but it is most often inapplicable in an urgent situation. Both urgent and definitive surgical management of post-tracheostomy arterial fistula are detailed in Chapter 27, “Repair of Tracheobrachiocephalic Artery Fistula.”7 Postoperative bleeding demands immediate exploration with an endotracheal tube, placed so that the cuff may be tightly inflated at the anastomosis to provide tamponade. The collar incision is reopened and complete sternotomy is added so that adequate exposure is obtained for proximal and distal control of the brachiocephalic artery. Cooper preferred upper sternotomy angled off into the right third interspace.8 This may help to avoid later sternal infection. Almost without exception, the artery is best excised, both arterial ends closed with running vascular sutures and covered with robust flaps of healthy tissue; that is, pedicled strap muscles, thymus, or omentum. If time permits, electroencephalographic monitoring can be helpful. Neurologic sequelae or subclavian “steal” syndrome becomes vanishingly rare after division of the brachiocephalic artery, as long as the carotid–subclavian
Tracheal Fistula to Brachiocephalic Artery
junction is intact.2,9 We have preferred not to perform arterial reconstruction in these circ*mstances since the field is contaminated and often infected. In a single case, a small arterial perforation was successfully excised, arteriorrhaphy done, and the site sealed with muscle. In general, the danger of a repeat hemorrhage is high after arteriorrhaphy, grafting, or simple ligation (versus suture closure) in this situation. If anastomotic separation has occurred after some days, tracheal reanastomosis is unlikely to succeed. The tracheal defect is initially spanned with an endotracheal tube to be followed by a tailored, long T tube when ventilation is no longer necessary. Later reconstruction may be possible. The tube is compartmentalized from the sutured arterial stumps by the viable tissue placed over them. The same principles of management, that is, arterial excision and tissue coverage, are applied to bleeding after cervicomediastinal exenteration, but the event is rare because of the prophylactic steps now advised (see Chapter 34, “Cervicomediastinal Exenteration and Mediastinal Tracheostomy”). The most likely source of massive hemorrhage following a carinal reconstruction is the pulmonary artery. Routine interposition of tissue over airway anastomoses has largely prevented this disaster (see Chapter 29, “Carinal Reconstruction”).
Results Wright summarized the literature to show 70 survivors of operations for tracheo-innominate artery fistulae but with only 40 surviving more than 2 months.10 Death is often related to the basic illness or its other complications. About 25% of patients who reach the operating room survive. As noted, neurologic problems are rare.
Prevention of a Tracheal-Arterial Fistula Prevention of hemorrhage after a tracheostomy is accomplished by 1) 2) 3) 4)
correct placement of the tracheostomy at the level of the second or third cartilaginous rings; avoidance of sharply angled (90˚) and excessively rigid tracheostomy tubes; checking the alignment of the tube in the trachea with a flexible bronchoscope at the completion of tracheostomy. It is always advisable to have a variety of tracheostomy tubes available; avoiding overinflation of tracheal cuffs. Check intracuff pressures routinely.
For prevention of hemorrhage after tracheal resection and reconstruction 1) 2) 3) 4)
do not dissect out the brachiocephalic artery unless necessary (adherence to trachea, prior surgery or dissection for tumor); interpose robust, vascularized tissue between the dissected artery and tracheal anastomosis (sternohyoid muscle, thymus, omentum); suture the sternohyoid muscle to the trachea over an artery if tracheostomy is anticipated, possibly later postoperatively; do not use prosthetic materials, especially in contact with major vessels.
For a description of prevention of a tracheal-arterial fistula in cervicomediastinal exenteration, see Chapter 34, “Cervicomediastinal Exenteration and Mediastinal Tracheostomy.”
References 1. 2.
3.
Gelman JJ, Aro M, Weiss SM. Tracheo-innominate artery fistula. J Am Coll Surg 1994;179:626–34. Jones JW, Reynolds M, Hewitt RL, Drapanas T. Tracheoinnominate artery erosion: successful surgical management of a devastating complication. Ann Surg 1976;184:194–204. Deslauriers J, Ginsberg RJ, Nelems JM, Pearson FG. Innominate artery rupture. A major complication of tracheal surgery. Ann Thorac Surg 1975;20:671–7.
4. 5. 6.
Levasseur P, Rojas-Miranda A, Kulski M, et al. Les complications de la chirurgie des sténoses trachéales nontumorales. Ann Chir Thorac Cardiovasc 1971;10:393–8. Grillo HC, Donahue DM, Mathisen DJ, et al. Postintubation tracheal stenosis. Treatment and results. J Thorac Cardiovasc Surg 1995;109:486–93. Grillo HC, Mathisen DJ. Cervical exenteration. Ann Thorac Surg 1990;49:401–9. Couraud L, Hafez A, Velly JF. Hémorrhages endotrachéales
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8.
par fissuration du tronc artériel brachio-céphalique au cours des canulations prolongées. Cure chirurgicale. Presse Med 1984;13:2577–9. Cooper JD. Tracheo-innominate artery fistula: successful management of three consecutive patients. Ann Thorac Surg 1977;24:439–47.
9. 10.
Brewster DC, Moncure AC, Darling RC, et al. Innominate artery lesions: problems encountered and lessons learned. J Vasc Surg 1985;2:99–112. Wright CD. Management of tracheoinnominate artery fistula. In: Mathisen DJ, editor. The trachea. Chest Surg Clin North Am 1996;6:865–73.
C H A P T E R F O U RT E E N
Infectious, Inflammatory, Infiltrative, Idiopathic, and Miscellaneous Tracheal Lesions Hermes C. Grillo, MD Infection Idiopathic Stenosis Inflammatory or Infiltrative Lesions Intrinsic Lesions Which Deform the Trachea
In addition to clearly defined and relatively more common diseases of the trachea (ie, primary and secondary neoplasms, postintubation lesions, congenital and traumatic lesions), a wide variety of uncommon conditions may be encountered. These lesions can be either intrinsic or extrinsic. Intrinsic lesions include the following: 1) those due to specific infection, such as tuberculosis and histoplasmosis; 2) defined conditions that affect other organs or tissues besides the trachea and bronchi, broadly characterized as inflammatory or infiltrative, but fundamentally of unknown etiology, including sarcoid, amyloid, Wegener’s granulomatosis, and relapsing polychondritis; 3) idiopathic laryngotracheal stenosis; and 4) a miscellany of intrinsic conditions that present as deformation or malacia of the trachea. Extrinsic lesions, which cause tracheal obstruction by compression, include goiter, tumors and cysts, congenital and acquired vascular lesions, and mediastinal or chest wall displacement. Most of these two types of lesions are covered in this chapter, except for malacia and extrinsic compression, which are presented in Chapter 15, “Tracheobronchial Malacia and Compression.”
Infection Tuberculosis Tracheobronchial tuberculous infections seem to involve principally the lower trachea and/or main bronchi (see Figure 37 [Color Plate 16]). Acute ulcerative tuberculous tracheitis is treated medically. Polypoid tissue and then cicatricial stenosis may result as the tracheitis or bronchitis heals.1 This can occur despite adequate treatment of the tuberculosis. Typically, the fibrosis that results is circumferential and submucosal (Figure 14-1). The airway may become extremely narrowed. Externally, the tracheal cartilages may appear to be intact, despite some peritracheal fibrosis. These days, cavitary disease is not often present with a tuberculous stenosis, as it once was. Primary parenchymal disease associated with endobronchial tuberculosis has been noted more in the lower lobes in the form of bronchiectasis. The length and severity of stenosis varies. The degree of resolution of
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the frequently accompanying pulmonary lesions also varies, ranging from residual parenchymal scar to lobar fibrosis or destruction (Figure 14-2). Tuberculous broncholithiasis is not often seen anymore. Rarer still are tuberculous tracheoesophageal fistulae.2 If possible, active tuberculosis should be arrested before surgical resection and reconstruction is contemplated. The linear extent of mature fibrous stenosis of the trachea and bronchi may be such that excision and reconstruction is not presently possible. This leaves the possibility of dilation and stenting. When the stenosis is more limited in extent, surgical excision and reconstruction can be performed, with the likelihood of a good result. Such resections have included excision of the lower trachea and carina, isolated resection of the left main bronchus, as well as sleeve lobectomies.3–5 Kato and colleagues, however, noted an increased frequency of postanastomotic stenosis.5 In 2 patients operated upon for acute and severe airway obstructions, the resection necessarily included a stenotic lower trachea, a stenotic right main bronchus, and a contracted and fibrotic right upper lobe. The right bronchus intermedius was anastomosed to the side of the trachea after the left main
FIGURE 14-1 Tuberculous stenosis of lower trachea and right main bronchus. The bronchial cross section (arrow) shows the lumen nearly obliterated by circumferential fibrosis.
FIGURE 14-2
Complete specimen from Figure 14-1, consisting of stenosed lower trachea and right main bronchus to the bronchus intermedius. The right upper lobe was also densely fibrosed and removed in continuity.
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bronchus had been joined end-to-end to the trachea (Figure 14-3). One patient with a fibrotic stenosis recovered well. In the second patient, with markedly active disease, the healing failed and the patient died. In the presence of active tuberculosis, obstruction must be managed other than by resection. This same distribution of disease and treatment has been described by others.5,6
Histoplasmosis Histoplasmosis causes airway obstruction in several ways.7 Large fibrotic or calcified lymph nodes may compress the main bronchi by forming what is well described as subcarinal histoplasmoma or mediastinal granuloma (Figure 14-4). The center of this mass usually contains necrotic material. The periphery is characterized by a considerable thickness of dense collagenous tissue. Enlarged lymph nodes and fibrosis may compress the right or left main bronchi, and, in particular, the bronchus intermedius, at the level of the large lymph node accumulation present around the middle lobe bronchus (Figure 14-5).7,8 Calcified lymph nodes may also erode gradually into the carina, the right or left main bronchi, or the bronchus intermedius, causing obstruction and hemoptysis from granulation tissue, as well as eventual protrusion of the calcific node (Figure 14-6). Histoplasmosis is now a principal cause of broncholithiasis, as tuberculous disease has receded (see Figure 36 [Color Plate 16]).9 Intrinsic fibrosis of the wall of the lower trachea and one or both main bronchi or of the right bronchial tree and bronchus intermedius may also occur, with accompanying lymph node involvement (see Figure 35, Color Plate 16). Varying degrees of mediastinal fibrosis can present (Figure 14-7). Pulmonary infection and fibrosis may follow bronchial obstruction, and hemorrhage
A
B
FIGURE 14-3 Tomograms (retouched) of a 38-year-old woman with tuberculous stenosis seen in the surgical specimen in Figure 14-2. A, Lower tracheal and right main bronchial stenoses are indicated by arrows. B, Postoperative study shows anastomosis of bronchus intermedius (arrow) to side of trachea just above the end-to-end anastomosis of trachea to left main bronchus.
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FIGURE 14-4 Magnetic resonance image showing bilateral compression of main bronchi by a subcarinal mass due to histoplasmosis in a 35-year-old woman. Extensive disease also encircles the bronchus intermedius. The lesion developed radiologically in less than 5 years. This required removal of middle and lower lobes in continuity with the subcarinal mass. The main bronchi were intact.
FIGURE 14-5 Bronchus intermedius constricted by dense scar which envelopes cartilages, causing them to override. Nodes at the origin of this bronchus are frequently involved.
may accompany broncholithiasis. The granulomatous process may compress the adjacent esophagus to some degree and even produce fistulae from the esophagus to the subcarinal or lobar lymph nodes or to the airway itself (see Figure 12-12 in Chapter 12, “Acquired Tracheoesophageal and Bronchoesophageal Fistula”).7,10 Inability to identify Histoplasma capsulatum from biopsy specimens in many patients has led to conclusions that fibrosis is often not the result of active fungal proliferation but instead of hypersensitivity reaction to the healing infection.11 Although not easily established, the interval between initial infection and these later presentations appears to be of several years. Patients present with cough (41%), dyspnea (32%), hemoptysis (31%), or recurrent postobstructive pneumonia (23%). Pleuritic pain also occurs (23%).11 Forty percent of patients may be asymptomatic. Superior vena cava syndrome is another presentation that may coexist with airway obstruction. Computed tomography (CT) scanning with contrast has been of particular help in identifying the extent of involvement (see Figure 14-7B). Skin tests are of no use, in view of the widespread sensitization of the population in areas where the disease is endemic (up to 80%). Indeed, skin tests may be misleading, by causing conversion of serologic tests. Serial complement fixation titers may be of value and may help to determine the need for antifungal therapy in chronic states.12 Antigenuria may be present in active disseminated infection. Organisms are most often demonstrated by silver methenamine stains in pathological material rather than found in aspirates, even in the presence of bronchial erosion. Although acute documented histoplasmosis is treated with amphotericin, the use of the drug has not proved to be of benefit in late cases of fibrosis without demonstration of active organisms. The finding of organisms may indicate the use of itraconazole or ketoconazole, which have less side effects than those seen with amphotericin. Fibrosis may be so severe and so dense that dilation finally becomes impossible (see Figures 14-7, 14-8). There may be no useful medical treatment at this stage. Large, obstructing subcarinal masses may sometimes be removed by painstaking surgical excision, without the necessity of bronchial resection.
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A
B 14-6 Mass of calcified subcarinal lymph nodes due to histoplasmosis, with broncholithiasis of the left main bronchus. A, Computed tomography scan showing a calcified mass. B, Tomogram of the carina in the same patient. The arrow points to the broncholith protruding into the bronchial lumen. C, The mass excised with an attached intrabronchial extension. The bronchus was closed longitudinally in this particular patient, which is not always possible. Also, see Figure 36 (Color Plate 16). FIGURE
C
A resulting bronchial opening, if very limited, may indeed be closed with only slight ultimate narrowing of the bronchus. Small portions of the fibrotic wall of “histoplasmoma” may be left in place in the mediastinum rather than incurring serious technical problems or creating an unreconstructible situation.7,8 James and colleagues essentially performed decortication of a severely stenotic trachea, which produced lasting relief despite necessarily incomplete removal of fibrosis.13 Garrett and Roper noted relief of vascular and bronchial obstructions by unroofing soft nodes, leaving the adherent fibrous capsule behind.14 In their series of 94 patients, 13 had respiratory symptoms, with major bronchial narrowing in 9 and compression in the other 4. Eleven patients had hemoptysis related to broncholithiasis. Postobstructive destruction of the middle and right lower lobe requires resection. The technical procedure may be very difficult and often requires judicious placement of a proximal tourniquet on the pul-
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monary artery prior to proceeding with further dissection. The calcified lymph nodes often intrinsically involve the wall of the pulmonary artery. We reported bronchoplastic procedures for airway involvement as follows: sleeve lobectomy in 3 patients, carinal resection in 1, carinal pneumonectomy in 4, and sleeve resection of the right main bronchus in 1.7 The organism was identified in 9 of 20 patients by staining. Despite the adjacent fibrosis, the tracheobronchial anastomoses heal if there is not excessive tension. Dense cicatrization may involve a proximal pulmonary artery, necessitating intrapericardial control. Full thickness esophageal involvement or fistula is managed by careful layered esophageal closure, with firm buttressing of the suture line. Significant collateral circulation from the pleura to lung, sufficient to cause severe hemoptysis, may occur, especially where a pulmonary artery is occluded.11 Superior vena cava obstruction is also well recognized in this process. Surgical intervention does seem to be advisable, even for asymptomatic mediastinal granulomas of large size. Dines and colleagues found that 34% of their cases of mediastinal granuloma progressed to
A
C FIGURE 14-7 Massive mediastinal fibrosis in a 57-year-old man who had reached an extreme of incapacitating dyspnea. Prior thoracotomy had failed and bronchial dilation proved impossible. A, Detail of chest roentgenogram showing critical stenosis of the right main bronchus (right arrow) and bronchus intermedius plus severe stenosis of the left main bronchus (left arrow). B, Computed tomography scan demonstrating a fibrocalcific mass at the carina in the same patient. C, Bronchoscopy reveals near total obstruction of the right main bronchus and a tiny opening on the left.
B
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D
E
F
FIGURE 14-7 (CONTINUED) D, Operative treatment. Right carinal pneumonectomy was necessary, with intrapericardial division of the pulmonary artery, since calcification extended almost to the origin of the right pulmonary artery. Exposure was via a right thoracotomy. Recovery was uneventful. E, Specimen shows calcific and anthracotic lymph nodes encased by dense fibrosis. The left main bronchus is at the left (arrow). F, Photomicrograph of dense keloidal fibrosis (hematoxylin and eosin stain). Many fibroblasts are seen on the left and thick bundles of collagen on the right. Histoplasma capsulatum was identified on silver methenamine stain.
fibrosing mediastinitis within 2 years, a result at variance with others.10,11,14 In a study of 71 patients, Lloyd and colleagues found no support for evolution of a mediastinal granuloma into mediastinal fibrosis.11 Trastek and colleagues advised surgical removal of broncholiths rather than bronchoscopic attempts at removal, since pathologic involvement so often necessitated pulmonary resection.9 We concur in this approach. The endoscopically visible broncholith is truly only the “tip of the iceberg.”
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Other Infections Nocardiosis was reported as a mass in the right main bronchus in an isolated case.15 A necrotizing mucormycosis that involves the trachea, carina, or bronchi, as well as the lungs, is seen principally in diabetic patients and in patients who are immunosuppressed or undergoing chemotherapy, particularly for lymphoma. Chronic renal failure and a history of organ transplantation are factors.16 Diagnosis is made on bronchial biopsy material by direct examination. Branching, nonseptate hyphae are noted with necrosis, fibrosis, and vessel thrombosis. Cultures are difficult to obtain. Progression of the infection from the bronchus may cause severe or fatal hemorrhage from pulmonary vessels.17 Secondary bacterial infection also occurs in the lungs. Prompt and very radical excision of the involved airway and lung, under the protection of vigorous and prolonged treatment with amphotericin or other drugs, may save some of these patients.16,18 In a young
A
B FIGURE 14-8
Intrinsic fibrosis of the airways often accompanies mediastinal processes due to histoplasmosis, as seen in Figure 14-7. Further examples are shown here. A, Tracheal tomogram showing narrowed trachea as well as mediastinal mass. Exploration had been previously done. B, Section of a densely fibrotic stenosed trachea in another patient. C, A carina similarly encased and invaded by fibrosis in another patient.
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diabetic woman with destructive mucormycosis in the right lung, extensive disease involving the carina and lower third of the trachea, and with infiltrates in the opposite lung, who was deteriorating on drug treatment, an aggressive right carinal pneumonectomy and lower tracheal resection, combined with continued medical treatment, led to resolution and complete recovery. Only a few instances of tracheal bronchial mucormycosis are reported.19 In a review of reported patients, Brown and colleagues confirmed the favorable results of early surgical treatment, against a high fatality with medical treatment alone.17 Tedder and colleagues compared a mortality of 68% for medical treatment versus 11% for surgical and medical treatments.16 Similar results were obtained by Donahue and Wain.18 Diphtheria in childhood may be followed many years later by tracheal stenosis or laryngotracheal stenosis; that is, involvement of the subglottic larynx as well as the upper trachea. Since most of the patients who had this disease in infancy or in early childhood were treated with one or more intubations and tracheostomies, it is difficult to determine whether the later stenosis is due to disease or to treatment. In several cases, the patient carried a tracheostomy for many years, later had it removed, and then appeared even later with severe stenosis. These late stenoses, which in my experience most frequently involve the subglottic larynx as well as the uppermost trachea and which lie in the region of prior tracheostomy, may be reconstructible. The anatomic and surgical considerations in such cases are the same as those for postintubation stenoses. Diminishing occurrence of these cases undoubtedly reflects the success of immunization programs. Scleroma (rhinoscleroma) is a rare disease, which may cause fibrosing changes in the nasopharynx, larynx, and upper airways. In only 2% of cases does it occur in the trachea and bronchi.20,21 Scleroma is related to infection by Klebsiella rhinoscleromatis. The organism is identified in biopsied material. The disease occurs in Mexico, Central and South America, Eastern Europe, the Middle East, India, and only occasionally in the United States. Scleroma is most common in the first three decades of life and in patients with poor nutrition. As the disease appears and progresses, it is characterized by nasal obstruction, nasal deformity, hoarseness, epistaxis, sore throat, and lip swelling.21 The tracheobronchial tree is kept open in these patients by repeated bronchoscopies, while prolonged and repeated antibiotic treatments (streptomycin and tetracycline) are given. A tracheostomy may become necessary. Glottic webs and subglottic scars may result as healing proceeds. Although viral in origin, laryngotracheal papillomatosis is described in Chapter 7, “Primary Tracheal Neoplasms,” because of its tumor-like appearance. Endobronchial Kaposi’s sarcoma is also described in Chapter 7. Human immunodeficiency virus (HIV)-infected persons are also subject to endobronchial tuberculosis, aspergillosis, non-Hodgkin’s lymphoma, and bacterial tracheitis.22
Idiopathic Stenosis Cicatricial stenosis with a lesser inflammatory component, localized in the subglottic larynx and upper trachea, occurs without known cause.23,24 This process is labelled idiopathic laryngotracheal stenosis (Figure 14-9). None of these patients have been intubated for ventilation or have suffered external or internal trauma to the trachea. The lesions are not congenital. There are no findings in this group of patients of associated mediastinal fibrosis or lymph node involvement by any pathologic process. Few patients have histories or findings suggesting esophageal reflux and aspiration. None have had specific or nonspecific tracheal infections, nor did they later develop manifestations of systemic disease such as polychondritis, or amyloid. A few with stenosis due to Wegener’s granulomatosis confined to the upper airway were initially misdiagnosed as idiopathic stenosis, prior to routine screening with an antineutrophil cytoplasmic antibody (ANCA) test. In a series of 73 patients, 71 were female. Age distribution varied widely from 13 to 74 years, but the disease was seen chiefly in the third, fourth, and fifth decades of life.24 Initial symptoms of dyspnea on effort (in 52%) progressed to dyspnea at rest, with noisy breathing, wheezing, or stridor (in 48%). The duration of symptoms prior to initial presentation varied between 4 months to over 30 years, with the greatest number reporting 1 to 4 years of symptoms. Careful history is necessary to identify the often subtle
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14-9 Diagrams of typical distributions of idiopathic laryngotracheal stenosis. A, The lesion often impinges on the low subglottic larynx at the level of the cricoid cartilage. A resection need remove only a small margin of the lower cricoid. B, A lesion that begins in the subglottic larynx and extends to varying distances into upper trachea. Narrowing usually begins shortly below the vocal cords, but enough space remains below the glottis for laryngotracheal resection and adequate primary anastomosis. C, In this case, the stenosis is more severe immediately below the vocal cords, with the lumen, even at that level, being very narrow and possibly inadequate for anastomosis. Reproduced with permission from Grillo HC et al.23 FIGURE
onset of the earliest symptoms. Patients were frequently misdiagnosed as having asthma. It is essential to obtain ANCA titer on every patient to rule out Wegener’s disease.25 Bronchoscopic biopsies are not diagnostic of either disease. Nasoseptal biopsies, on the other hand, may identify Wegener’s granulomatosis histologically. Prior to the 1993 report by Grillo and colleagues, only limited or individual case reports were available.23 Koufman, in a detailed study of the laryngeal effects of gastroesophageal reflux, noted that 30% of otolaryngologic patients had pharyngeal reflux, as determined by a double pH probe technique.26 Seventyeight percent of patients with a laryngeal stenosis from any cause, but principally of postintubation origin, showed pharyngeal reflux. Maronian and colleagues, using three to four port pH probes, with one positioned proximal to the upper esophageal sphincter, recorded a pH of less than 4.0 in 5 of 7 patients with isolated idiopathic subglottic stenosis.27 No control data were presented, however, as has most often been the case in assessing the significance of laryngeal gastroesophageal reflux. The significance of these findings as indicators of possible cause for idiopathic stenosis remains uncertain. Fifteen of our patients had a history or symptoms of gastroesophageal reflux. It must be noted that none of our patients who were operated upon for idiopathic stenosis suffered later progression of stenosis.24 This would have been expected if untreated reflux was an important etiologic factor. In most cases, radiologic study showed a circumferential lesion of varying length, most often between 1 to 3 cm, centered at the junction between the cricoid cartilage and trachea (Figure 14-10). In 4 cases, the subglottic larynx itself was not involved, but in another 69 cases, subglottic involvement was of varying degrees of severity. The narrowing most often began shortly below the vocal cords and rapidly became a more severe to maximal stenosis at approximately the cricoid level. Although the involvement might be eccentric, it was always circumferential. Vocal cord function appeared normal. Longitudinal roentgenograms brought out the extent of the lesion and its nature most clearly. Flow volume loops, as expected, demonstrated extrathoracic fixed obstruction. Cultures obtained from biopsies and from surgical specimens showed nothing but the usual upper respiratory flora. Skin tests and serologic tests, done
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B FIGURE 14-10
A
Images of upper airway of a 50-year-old woman with typical idiopathic laryngotracheal stenosis. A, Tomogram of the larynx and upper trachea. The false and true vocal cords are well outlined at the top. The stenosis involves the subglottic larynx and uppermost trachea. Compare with Figure. 14-9B. B, Computed tomography scan of the neck. The circumferential disposition of the stenosis lies within the ring of cricoid cartilage. Occasionally, the lesion is eccentric, although still circumferential.
especially in the earlier patients, were negative for tuberculosis, coccidioidomycosis, histoplasmosis, and blastomycosis. On bronchoscopy, the mucosa over the lesion appeared to be injected, and bled easily. Granulation tissue was uncommon (Figure 14-11). Ulceration was identified in only 1 patient. When these patients presented, the diameter of the aperture ranged from 3 to 10 mm, with most lying between 5 to 7 mm. The trachea distal to the stenosis appeared normal (see Figure 33 [Color Plate 15]). Since the origin of a laryngotracheal and upper tracheal idiopathic stenosis is not understood, and since the entity had not been previously studied or followed over a long period of time, my initial approach was conservative. Some lesions remained stable over the period of observation, but in others, the obstruction worsened, requiring frequent dilations. Linear extension was not seen, however. Seventy-three patients have been subjected to surgical resection and reconstruction, with 4 cases involving the upper trachea, and in 10 cases, a rim of lower cricoid cartilage as well. In 59 cases with subglottic laryngeal stricture as well, the anterior portion of the subglottic larynx was removed and the posterior portion of the stenosis was managed by laryngotracheoplasty, as described in Chapter 25, “Laryngotracheal Reconstruction” (Figure 14-12). Thirty-six of these 59 patients required posterior laryngeal cricoid resurfacing with membranous tracheal wall. Initially, a protective complementary tracheostomy was performed in patients requiring laryngotracheoplasty. However, this was soon found to be unnecessary as a routine measure. Seven of 73 patients had temporary tracheostomies, but of the last 30 patients, only 1 required this. In 2 patients, in whom the stenosis was extremely severe and extended upward to the undersurface of the vocal cords, laryngofissure, excision of the scar, and resurfacing with buccal mucosa were performed. In 72 patients
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14-11 Bronchoscopic findings. A, View through separated vocal cords, demonstrating the funnelled subglottic larynx, with the circumferential ring of a high idiopathic stenosis visible below. Correction required circumferential laryngotracheoplasty as described. B, Closer intralaryngeal view affirms the circular nature of the stenosis. Its short length and the normal tracheal rings below are well seen. No granulation tissue is present but the mucosa is easily abraded. Also, see Figure 33 (Color Plate 15).
FIGURE
14-12 Laryngotracheal resection and reconstruction for idiopathic stenosis, in a 55-year-old woman with a 3-year history of progressive symptoms. A, Preoperative tomogram. The solid arrow indicates the glottis; the open arrow indicates the stenosis. B, Postoperative tomogram. The arrow is at the glottis. Vocal cords are symmetrical. Note the slight indentation at the level of the anastomosis. The patient continued to do well 22 years after the operation. FIGURE
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with proximal idiopathic laryngotracheal stenosis, who underwent operation and had long-term follow-up (median 8 years), 19 (26%) attained excellent results in respect to voice and airway, 47 (64%) had good results, and 5 had only fair results and required occasional dilation (Figure 14-13). One case with poor result required at least annual dilation. In only this patient was there evidence of extension of the inflammatory process beyond that originally seen, suggesting a different process. Of the patients followed conservatively, none demonstrated spontaneous regression or regression in response to other treatments, including systemic or local corticosteroids. In light of our observations in 73 patients of 1) long-term good results from surgical treatment in most, and 2) failure of recurrence or progression of the disease, it is difficult to understand the contradictory results of Dedo and Catten, who reported relentlessly progressive disease and complete failure in their 7 patients treated surgically.28 Pathologically, fibrosis was generally circumferential and of even thickness. Preeminent was dense collagenous fibrosis of keloidal type, which thickened the lamina propria of the trachea (Figure 14-14).23 Fibroblasts were relatively sparse. The surface epithelium usually showed squamous metaplasia. Inflammation was not prominent. Granulation tissue was also seen. Sometimes, this might well have been related to prior dilations, lasering, or other treatment. Cartilaginous rings were intact and essentially normal. There were no histologic characteristics to suggest a relapsing polychondritis, vasculitis, Wegener’s granulomatosis, or amyloid, and calcification or stainable organisms were not seen. The location, configuration, gross appearance, and microscopic appearance of these upper airway stenoses are similar enough to suggest a definable disease entity. The predominance of the disease in females deserves notice. In idiopathic lesions that involve the subglottic larynx (and these are in the majority), airway narrowing usually begins shortly below the vocal cords. It takes refined and experienced judgment to decide which patients should be operated upon. A reasonably sized “atrium” is needed below the vocal cords to provide an adequate luminal size for successful anastomosis. It should allow a luminal cross-sectional area of the airway, at least 50% of normal. The obliquity of the anastomosis helps to enlarge the new laryngotracheal junc-
14-13 Functional results of reconstruction. Flow volume loops before and after operation, showing marked improvement in inspiratory and expiratory flows. Reproduced with permission from Grillo HC et al.23
FIGURE
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FIGURE 14-14 Pathologic findings in idiopathic stenosis. A, Gross specimen, cross sections of trachea. The dense fibrosis lies inside of intact cartilaginous rings. B, Photomicrograph reveals keloidal fibrous tissue, which replaces the lamina propria of the tracheal mucosa. Inflammatory changes are found in many near the mucosal surface, and frank granulation tissue in a few (hematoxylin and eosin; ×125 original magnification).
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tion. Contraindications to operation are a significant stenosis that begins within 5 mm of the glottis and, less permanently, the presence of florid inflammation and/or granulation tissue in the stenosis. If the stenosis reaches or nearly reaches the undersurface of the vocal cords, then treatment by periodic dilation may be the best option. If florid inflammation and granulation tissue are present, the operation should be deferred. Periodic mechanical dilation (see Chapter 19, “Urgent Treatment of Tracheal Obstruction”), with or without local corticosteroid injection, permits temporizing for as long as is necessary. Operative failure here would likely be complete and permanent, with scant opportunity for a second repair. Furthermore, the recurrent laryngeal nerves are at risk in all of these patients. Results are also limited in many patients by a permanent slight weakness in the ability to project voice and in a diminished ability to sing, which are common sequelae (42 of 67 patients with good to excellent surgical results) after surgical reconformation of the larynx. In a much smaller number of patients, a different stenotic process, also idiopathic, was encountered at the supracarinal and carinal levels, which also involved the main bronchi (Figure 14-15). In another small number of patients, a more inflammatory stenosing process would usually (but not always) spread over time, to involve almost the entire trachea and main bronchi (Figure 14-16). In view of the persistently anatomically localized nature of the upper idiopathic tracheal stenoses, it seems likely that these few cases
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C FIGURE 14-15 Idiopathic stenosis of the distal trachea and carina in a 62-year-old woman with a 10-year history of “asthma,” surgical correction of a high tracheal stenosis, with finding of carinal stenosis a year later, the duration of which is unknown. A, Tracheobronchogram showing stenosis of both main bronchi (arrows). Oblique view. B, Bronchoscopic view of the carina in this patient. C, Surgical specimen. Trachea was anastomosed endto-end to the left main bronchus. The right main bronchial stump was implanted in the right wall of the trachea above the other anastomosis. The result remained good.
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of more diffuse and sometimes progressively longer stenoses are of other etiology or etiologies. These few patients did not show clinical or pathological characteristics of histoplasmosis.
Inflammatory or Infiltrative Lesions A group of unrelated lesions are discussed here. Their only common feature is an alteration or infiltration of tracheal and bronchial walls by non-neoplastic processes that are clinically well characterized, but are of unknown cause. Idiopathic stenosis could well be included, in that it largely shows collagenous deposition in the larynx and trachea.
Relapsing Polychondritis Relapsing polychondritis remains a disease of unknown origin, characterized principally by inflammatory degeneration of articular and extra-articular cartilages. It is believed to be an autoimmune disease, possibly a reaction to type II collagen, and has been reported in association with Wegener’s granulomatosis, rheumatoid arthritis, vasculitis, and systemic lupus erythematosus.29 The clinical course may be rapid or slow, and episodic or progressive.30 Cardiovascular, renal, and neurologic manifestations occur later. A variety of other systemic diseases have been seen in association as well.29 In those with a fully developed syndrome, the cartilage
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FIGURE 14-16 Other examples of unusual lower tracheal and carinal stenoses of unknown origin. A, Bronchoscopic view in a 72-year-old woman with a 15-year history of progressive dyspnea. Successfully treated by carinal resection, anastomosis of the trachea to the left main bronchus, and implantation of the right main bronchus into the side of the trachea. B, Surgical specimen from a 55-year-old male with stenosis of the lower trachea and carina. Dense collagenous fibrosis is evident. C, Idiopathic stenosis of the left main bronchus in a young woman. After initially good result from a left main bronchial resection, she later developed progressive stenosis from the carina upward. The entire trachea became involved and she has been maintained by periodic dilations. A similar patient eventually died from obstruction.
of the nose deteriorates, leading to a saddle nose, cartilages of the ears become thickened and inflamed, and cartilages of the airways from the larynx to the segmental bronchi may be involved. McAdam and colleagues collected reports of 159 patients with relapsing polychondritis, including 29 studied prospectively.31 In addition to the elements noted, these patients had inflammatory polyarthritis, nasochondritis, ocular inflammation, and cochlear and vestibular ear injuries. As the cartilage is destroyed by the recurring inflammatory process, it is replaced by fibrous tissue. Initial airway obstruction is due to edema and inflammation before the cartilage is destroyed and before airway collapse occurs. Fever, weight loss, and lethargy accompany the illness. The onset of symptoms is most frequently in the fourth decade of life, but may be seen much earlier. Initial presentation may be with hoarseness, loss of voice, and tenderness over the larynx and trachea. The larynx and proximal trachea are most commonly affected.32 The interval between first airway symptoms and
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declaration of the disease’s characteristics may be prolonged. Age range at diagnosis is from 13 to 84 years, but most are between 44 and 51 years of age. Men and women are affected equally.29,31 Fifty-six percent of patients had evidence of respiratory tract involvement.31 Eleven of 14 patients who presented initially with respiratory involvement required a tracheostomy. An additional 58 patients later developed respiratory problems, and 15 of them required a tracheostomy. Although the edematous and inflammatory processes may respond to cortisone treatment, cartilaginous destruction continues. In McAdam’s series, 13 patients died of airway collapse or obstruction, and 4 more of pneumonia. The histologic picture is not absolutely characteristic of any disease but may be suggestive. Specific diagnostic tests are lacking. In a review of experience with 36 patients and study of 30 additional patients in 1998, Trentham and Le found a similar prominence of auricular chondritis (92%), arthritis (48%), laryngotracheal symptoms (39%), nasochondritis (33%), and ocular inflammation (25%), but in greater percentages than in earlier series.29 Audiovestibular problems occur in 6 to 9%. Delay between first symptoms and diagnosis averaged 2.9 years. Radiography, including tomography, conventional and computed, show upper airway changes and the latter lower airway changes (Figure 14-17). Dynamic studies may demonstrate collapse more clearly. Pulmonary function tests, especially flow volume curves, offer a means of following the disease progression. Bronchoscopy, or any manipulation of the airway, must be done with a light touch to avoid inciting further edema, inflammation, and acute obstruction. In addition to treatment with corticosteroids, cytotoxic agents (such as cyclophosphamide, methotrexate, and azathioprine) are used. Beyond tracheostomy, and a T tube for temporary relief in a few with upper airway disease, there is no standard surgical treatment. The disease is too extensive and progressive to be managed by resection or reconstruction. When the larynx is severely involved, a tracheostomy becomes necessary. If the disease progresses distally to involve the lobar and segmental bronchi, there is lit-
14-17 Relapsing polychondritis. Tracheal tomogram in a 28-year-old male with progressive dyspnea on exertion. The trachea is diffusely narrowed to 12 mm from just below the cricoid to carina. The left main bronchus is also narrowed to 6 mm. Over 12 years, the narrowing worsened. Computed tomography scan confirmed circumferential narrowing, thickening, and calcification of tracheal or bronchial walls. Subsequently, ear changes occurred and biopsy of cartilage was consistent with relapsing polychondritis.
FIGURE
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tle that can be done therapeutically at present. Pulmonary sepsis may follow diffuse bronchial collapse. Survival with medical treatment has improved some over the years.29
Sarcoid Sarcoid (or sarcoidosis) is a systemic disease of unknown origin or origins, possibly an autoimmune condition. It demonstrates racial proclivity for blacks. Many organ systems may be involved, but almost always the respiratory system. The condition is characterized pathologically by noncaseating granulomas. Sarcoid varies in manifestations, severity, and outcome.33 In addition to nonspecific symptoms such as fatigue, anorexia, weight loss, and fever, respiratory complaints may include exertional dyspnea, retrosternal chest pain, and cough. Respiratory symptoms are prominent, but many patients are asymptomatic. The full spectrum of the disease will not be recounted here. The thoracic surgeon’s encounter is often in reply to the need for mediastinal lymph node biopsy or bronchoscopic carinal biopsy, since both tissues are highly susceptible to sarcoidal granulomatous involvement. Nearly half of the bronchoscopic biopsies and more of the mediastinal node biopsies will be positive, but other causes of noncaseating granuloma must be ruled out. Sarcoid produces major airway obstruction in two ways: 1) by massive enlargement of mediastinal and hilar lymph nodes, with compression and distortion of the airway; and 2) by intrinsic fibrotic change in the wall of the trachea and bronchi (Figure 14-18).34 Endobronchial nodules may be present with or without visible or symptomatic bronchostenosis.35 Sarcoid may also cause hoarseness and, later, obstruction by laryngeal involvement. These changes are usually concomitant with parenchymal pulmonary changes. Airflow limitation, wheezing, and stridor may be present in 10% of patients, although the usual functional defect in pulmonary sarcoid is restrictive.35 When the walls of the trachea or main bronchi or both of these structures are involved, the length of the stenotic segment may be long and the process progressive. Because of the diffuseness of involvement and progression, these lesions are rarely amenable to resection and reconstruction. Periodic dilation will tide some patients over for a long time. As the tissues contract by cicatricial evolution of the scarring, the hilum may be pulled upward and the left main bronchus, in particular, takes on a sharp curvature beneath the aortic arch, making continued dilation increasingly difficult. If both main bronchi are stenotic, the patient’s obstruction will mimic a fixed upper airway obstruction clinically and on functional study.36 Sarcoidosis may also cause airway obstruction in the larynx by granulomas. This is usually associated with cutaneous disease, especially of the lupus pernio form.37 I prefer to use a rigid bronchoscope with Jackson bougies, or a small diameter Maloney bougie to dilate a markedly angulated bronchus. Dilation has also been done via a flexible bronchoscope.38 There is no clear proof of the efficacy of inhaled corticosteroids, or, for that matter, systemic steroids for major tracheobronchial obstruction.
Wegener’s Granulomatosis Wegener’s granulomatosis is a disease of unknown etiology characterized by granulomas, vasculitis, and necrosis, which involves the upper and lower respiratory tract, kidneys, central nervous system, and other organs. In 158 patients, 97% were white, the genders were equal in number, and 85% were over 19 years of age.39 Typically, patients suffer serious consequences of the disease or its treatment (cyclophosphamide and glucocorticoids). In 99 patients followed longer than 5 years, 44% had prolonged remissions, but 13% died of the disease or treatment. Methotrexate has served as an alternative to cyclophosphamide and other drugs will undoubtedly be used in the future.40 Sixteen percent of 158 patients treated at the National Institutes of Health (NIH) had subglottic stenosis, about double the previously reported incidence.41 Subglottic stenosis is more common in Wegener’s patients who are under 20 years of age. Some had limited disease, in that the kidneys were not involved. The larynx alone may be involved without evidence of vasculitis in other systems.39,42 Patients with subglottic stenosis from Wegener’s granulomatosis present with symptoms of effort
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B FIGURE 14-18 Intrinsic bronchial stenosis due to sarcoidosis. A, Tomogram of carina in a 30-year-old man with a 3-year history of repeated and persistent pneumonia in the right lung, with collapse and fibrosis of middle and lower lobes and air trapping in the upper lobe. The right main bronchus is severely narrowed and cut off distally, approximately where the upper lobe bronchus and bronchus intermedius branch. This patient also had episodes of left-sided pneumonia. B, Computed tomography scan in the same patient as A. The carina is displaced to the right. Note the patchy infiltrates in the left lung. The entire right bronchial tree was stenosed, and involved nodes were prominent in right paratracheal and subcarinal distributions. Pneumonectomy was necessary. C, Bronchoscopic view in another patient, showing severe left main bronchial stenosis.
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dyspnea, hoarseness, cough, and discomfort in the throat. The local lesion is best defined by linear laryngotracheal x-rays supplemented by CT or magnetic resonance imaging and by bronchoscopy (Figure 14-19). Laryngeal biopsies may not be diagnostic. Adequate nasoseptal biopsies, however, are often helpful.43 The ANCA is usually elevated and can be highly specific in diagnosis.25 In isolated lesions, the differential from idiopathic stenosis and from early polychondritis may be unclear (see Figure 34 [Color Plate 15]). Lebovics and colleagues found that 92% of 158 patients with Wegener’s granulomatosis had otolaryngologic manifestations, 25 with subglottic stenosis.41 Five responded to cytotoxic and steroid treatments alone. Sixteen of 20 who had fixed subglottic stenosis were treated by dilation, laser resection, and laryngotracheoplasty. Thirteen needed a temporary tracheostomy. Five underwent laryngotracheoplasty, 2 with microvascular reconstruction, using a rib graft with attached intercostal artery. An anterior and posterior cricoid split with cartilage augmentation was performed, with postoperative T tube stenting. Nonoperative treatment was applied first, including repeated intralesional injections of Depo-Medrol. Laser resection appeared to produce more scarring.
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A FIGURE 14-19 Wegener’s granulomatosis involving the upper airway. A, Anteroposterior tomogram of larynx and upper trachea. Note the severe narrowing of the subglottic larynx, beginning just below the glottis and extending into the proximal trachea. B, Lateral tomogram of the same lesion of A. C, Bronchoscopic view of the upper trachea in another patient. Irregular circumferential scarring is noted. In other cases, the gross appearance is similar to idiopathic stenosis. Bronchoscopic biopsy is not usually diagnostic. Also, see Figure 34 (Color Plate 15).
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Daum and colleagues from the Mayo Clinic found that 30 of 51 patients (59%) with proven Wegener’s granulomatosis, who were followed bronchoscopically, had endobronchial abnormalities due to the granulomatous processes.42 Five had subglottic stenosis, 18 had ulcerating tracheobronchitis (some with pseudotumors), 4 had tracheal or bronchial stenosis, and 2 had bleeding from unidentified sources. Seven
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of 9 patients with ulceration progressed to stenosis. Treatment was by dilation, laser, and Silastic stent. No correlation was found between observed inflammation and ANCA titers. Herridge and colleagues had success in 3 patients, who were in remission, treated by resection and thyrotracheal anastomosis, despite concurrent use of prednisone and cyclophosphamide.44 Protective tracheostomy or a T tube was used in all 3. Stenosis did not recur in 2 with longer follow-up. We performed resection in 6 patients with Wegener’s granulomatosis confined to the upper airway but misdiagnosed as idiopathic laryngotracheal stenosis. Two did well in long term, but 4 later restenosed. An NIH protocol from 1991 consisted of dilation and intralesional steroid injection at monthly intervals. The hope was that collagenous scar would be resorbed. Following dilation, the area of stenosis was injected with methylprednisolone acetate in four quadrants. One cc containing 40 mg of the drug was injected in each site. About half the volume leaked out. The patient was given Decadron briefly to minimize postoperative laryngeal swelling. Cyclophosphamide and glucocorticoids were continued only in those patients with other systems involved. Twenty patients underwent 113 procedures in total, with 14 requiring multiple treatments.45 Six patients with tracheostomies were decannulated and none of the others needed a tracheostomy, in contrast to earlier experience, where over half of the patients required a tracheostomy. Given the unpredictable course of Wegener’s granulomatosis, its varied manifestations, and response to treatment, conservative initial management of subglottic stenosis seems advisable. We presently employ repeated dilations with intralesional injection of methylprednisolone acetate (Depo-Medrol). Surgical resection is reserved for nonresponding patients who have a reasonably localized disease and who have been in prolonged systemic remission, and further, where the airway lesion does not evince florid inflammation. One-stage laryngotracheal resection is performed, when feasible (see Chapter 25, “Laryngotracheal Reconstruction”). Airway stenosis due to Wegener’s granulomatosis should be approached surgically with the greatest caution, if at all.
Amyloidosis Falk and colleagues point out that “amyloidosis is not a single disease, but a term for diseases that share a common feature: the extracellular deposition of pathologic insoluble fibrillar proteins in organs and tissues.”46 Different proteins make up the amyloid fibrils in primary amyloidosis (AL), in reactive systemic (secondary) amyloidosis (AA), and in rare familial disease (ATTR most common). Secondary amyloid has diminished in frequency with a lower incidence of chronic infectious diseases. Clinical presentations vary, but the organs most often involved are the kidney, heart, and peripheral nervous system. The disease is uncommon and airway involvement is more so. The now differentiated types of amyloid have not been identified in airway cases until recently. AL amyloidosis appears to have special affinity for lung tissue, presenting as nodular pulmonary and tracheobronchial amyloidoses.47,48 The hyaline-eosinophilic material also deposits in the lamina propria of the bronchial mucosa of different parts of the bronchial tree. Multiple coalesced nodules can narrow the bronchial lumen, and involvement ranges from a localized segment to a large part of the bronchial tree (Figure 14-20). Systemic amyloidosis, which often involves the lungs, has a poor prognosis. The pulmonary involvement is usually diffuse and infiltrative in this case. More localized, tumor-like amyloidoma, whether in the lung or tracheobronchial tree, appears to have a quite benign course, except for the consequences of airway obstruction, which may be fatal.48,49 It is largely dissociated from systemic disease. Tracheobronchial lesions may be quite localized or infiltrate over long distances of the larynx or bronchi.47,50,51 Multifocal submucosal plaques are more common than “amyloidomas,” or tumor-like masses. Regional lymph nodes may exhibit amyloid.48 In extensive tracheal involvement, amyloid material is also found, deposited peritracheally in the mediastinum and in the esophageal wall. Calcification may be noted in nodular amyloidomas on CT scan. In looking at 48 patients with lower respiratory tract amyloidosis, Hui and colleagues counted 28 with single or multiple nodules, 14 with tracheobronchial disease, of whom 4 were localized and 6 had diffuse interstitial pulmonary infiltrates.51
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B 14-20 Amyloidosis involving the airway. A, “Amyloidoma” localized to left main bronchus. The lesion fills the bronchus like a cork. A left main bronchial sleeve resection was performed, saving the lung. B, Anteroposterior view of the trachea, showing extensive linear involvement in a different patient. C,D Computed tomography scans of another patient, 54 years old, with tracheobronchial amyloidosis. The cervical trachea is markedly obstructed (C). The upper mediastinum and trachea in this patient are infiltrated by amyloid (D). Note the calcification. FIGURE
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14-20 (CONTINUED) E, Amyloid is present in the same patient’s tissues at the carinal level. Bronchi are thickened and calcified. F, Bronchial and mediastinal infiltration and calcification of amyloid are particularly well seen in the same 54-year-old patient. G, Tomogram of the larynx and trachea in a 19-year-old male with at least 10 years of slowly progressive dyspnea, much worsened in the year and a half prior to study. The upper arrow indicates the glottis. Below this is the image of the amyloid with calcification invading the larynx and trachea. The lower arrow indicates deformation of the midtrachea by mediastinal amyloid accumulation. FIGURE
A literature review in 1972 produced 25 instances of primary localized tracheobronchial amyloidomas: trachea only in 2, bronchus only in 9, and trachea and bronchus in 14.52 The genders were evenly spread, and the age range was 29 to 74 years (mean 53 years). Symptoms and signs were obstructive. Utz and colleagues described 4 patients with localized tracheobronchial amyloid in 55 patients with pulmonary amyloidosis (systemic and localized).49 Symptoms of nodular or tumorous tracheobronchial amyloidosis are cough, sputum, dyspnea, hemoptysis, and fever. The mucosa bleeds easily, but biopsy is necessary for diagnosis. Since it occurs so
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14-20 (CONTINUED) H, Computed tomography image of the same patient in Figure 14-20G, showing severe tracheal compromise. Note the calcification in the amyloid mass.The splayed shadow anterior to the trachea is the thyroid. I, Upper thoracic trachea is also invaded. Amyloid infiltrates the esophageal wall and narrows its lumen. A 5 cm length of the trachea and the anterior subglottic larynx were resected, plus esophageal muscular coats. Putty-like amyloid infiltrated the mediastinum and behind the posterior cricoid. Good result was finally achieved but a re-resection of the anastomotic stenosis, due to failure of healing of the first anastomosis, was required. FIGURE
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infrequently, it can easily be mistaken for a tumor, or even other infiltrative processes such as tracheopathia osteoplastica. Tracheobronchial amyloidosis is not usually associated with diffuse interstitial pulmonary amyloid deposition, as noted above. Classically, the protein stains with Congo red, but sophisticated techniques now permit precise determination of the amyloid type.46 Depending upon the location and extent of the process, surgical treatment may be possible.48,50 A localized tracheal or bronchial deposit may simulate tumor. The lesion may enlarge slowly, in time producing obstructive recurrent pneumonia or atelectasis. Death occurs from recurrent pneumonia or respiratory failure under these circ*mstances. If possible, the involved segment of trachea or bronchus is resected (see Figure 14-20A). Residual amyloid may necessarily be left at resection margins, predicting possible slow future recurrence. The rate of clinical progression is not defined, due to lack of experience. In one of our patients, a pulmonary shadow had been present for 22 years.48 In another very extensive lesion in a
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young man, an extended laryngotracheal resection was necessary (see Figures 14-20G,H,I). A T tube was required after partial anastomotic separation had occurred, but eventually, a re-resection of the localized stenosis was successfully accomplished, resulting in an intact airway despite the extent of resection. Naef and colleagues operated on a similar patient and cited 22 prior reported cases with diffuse or localized pseudotumors of the tracheobronchial tree, usually treated at that time by bronchoscopic means.50 In a diffuse disease, we have used palliative bronchoscopic core-out treatment, whereas others have preferred laser therapy.49 See also Figures 3-59, 3-60, and 3-61 (Color Plate 8).
Intrinsic Lesions Which Deform the Trachea A number of conditions largely display gross deformation of the trachea in several characteristic patterns. These changes appear to be intrinsic to the trachea rather than being due to compression by adjacent structures or masses. Included are saber-sheath trachea, tracheopathia osteoplastica, and tracheobronchomegaly.
Saber-Sheath Trachea A normal adult trachea has an essentially oval cross section, with the coronal diameter greater than that of the sagittal, although in many it is nearly circular (see Chapter 1, “Anatomy of the Trachea”). With increasing age, the configuration of the lower third of the trachea may be altered by the left lateral impression of an enlarging aorta. “Saber-sheath” trachea, however, describes a coronal narrowing of the entire intrathoracic trachea from both sides, with a corresponding increase in the sagittal diameter, usually affecting about the lower two-thirds of the trachea. The proximal cervical segment retains a normal shape (Figure 14-21). Sabersheath trachea is identified most often in older patients, particularly in males over 50 years of age. Ninetyfive percent of patients appear to suffer chronic obstructive pulmonary disease (COPD).53,54 Not all patients with COPD, however, show saber-sheath deformity. In most patients, it is an incidental finding. However, with extreme progression of the deformity, a large part of the posterior lateral walls of the trachea approximate to one another on coughing and on increased respiratory effort, especially expiratory. As the crosssectional area of the trachea is reduced, the rate of airflow is also reduced.54 The patient finds it increasingly difficult to generate a cough sufficiently forceful enough to clear tenacious secretions, which are characteristic of the pulmonary disease. Greene and Lechner described 13 patients with such narrowing, recalling the original use of the term in 1905.53 Patients’ ages ranged from 52 to 75 years, and clinical diagnoses were varied. All the patients had a history of heavy smoking, 10 had a diagnosis of chronic bronchitis, and 7 suffered from primary chronic obstructive pulmonary disease. The coronal intrathoracic tracheal diameters ranged from 7 to 13 mm with a mean of 10.5 mm; in contrast, the sagittal diameters showed a mean of 23.5 mm. The extrathoracic trachea was normal, with a coronal diameter of 20.6 mm. Ten of the 13 patients demonstrated calcification in the tracheal rings, which was probably age related. The mechanical forces that lead to this deformation of the intrathoracic trachea are not understood. It should be emphasized that the cartilages are not malacic. It is the change in shape and approximation on respiratory and tussive efforts that can lead to clinical problems. In a very rare case, I found it necessary to splint the trachea externally (using specially made polypropylene rings), in order to allow the patient to clear secretions adequately. A large bore tracheal T tube or inlying stent could also provide a means for maintaining an open airway during expiration and cough in these few critical patients. Definition of the extent to which the airway is obstructed by the process is obtained by bronchoscopic examination in the awake patient, flow volume curves, and inspiratory and expiratory thin-section CT scans of the trachea. Patients who have this deformity, but who are not symptomatic from it, and who require tracheal resection between the differently shaped proximal and distal portions of the trachea, do not present any difficulties in anastomosis. Sutures are placed proportionally. The tracheal ends are sufficiently flexible to permit easy anastomotic accommodation.
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14-21 Saber-sheath trachea. A, Anteroposterior (left) and lateral (right) roentgenograms, showing normal appearing cervical trachea with side-to-side narrowing of the intra-thoracic trachea. Near the carina, the trachea begins to widen again. FIGURE
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Tracheopathia Osteoplastica In tracheopathia osteoplastica (TPO), multiple submucosal osseocartilaginous nodules are present in the trachea, subglottic larynx, and bronchial tree (see Figures 42 and 43 [Color Plate 16]). An alternative name, tracheobronchopathia osteochondroplastica, is more completely descriptive, lacking only “laryngo”—but is excessively long and something of a tongue twister. The nodules are distributed only over underlying cartilages and not over the membranous wall. The most prominent nodules are usually seen in the trachea and the next most prominent in the main bronchi. Some are tiny and scattered like seeds. Others grow to considerable size (4 to 6 mm). The nodules are of hyaline cartilage with foci of ossification and lie submucosally, essentially forming a partial intratracheal osseocartilaginous cylinder (except over the membranous wall). Bridges of bone, cartilage, and collagen connect the inner tracheal cylinder formed by the pathologic bone and cartilage with the perichondrium of the regular tracheal cartilages. These findings led Young and colleagues to support ecchondrosis of the tracheal cartilages as the pathological process.55 An often present saber-sheath configuration in these patients may commence just below the cricoid cartilage, rather than being confined to the intrathoracic trachea, as it is in isolated saber-sheath tracheal deformation. Portions of the trachea may be more severely affected by TPO than others, and occasionally involve only part of the trachea. The etiology is unknown. Earlier suggestions that TPO is an end stage of amyloidosis have not held up.56 Other theories relate the calcifications to chronic purulent tracheitis or abnormalities in tracheal elastic tissue.56,57 This rare condition is seen in older adults and is often asymptomatic. Fifteen patients were diagnosed with TPO over 36 years at the Mayo Clinic.56 A number of cases reported were discovered incidentally at postmortem examination.55 TPO may, however, progress insidiously to produce respiratory symptoms, including dyspnea, cough, sputum production and retention, wheezing, hoarseness, and hemoptysis.56 A long history of exertional dyspnea and respiratory infection may be elicited. As obstruction worsens, the patient has progressive difficulty in raising viscid secretions.
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FIGURE 14-21 (CONTINUED) B, Computed tomography scans showing changes in tracheal configuration with progression, from the cervical (left upper image), to the upper thoracic (right upper and left lower image), to the lower thoracic (right lower image). The shape of a saber sheath is best exemplified in the right upper image. As the carinal branching is reached, the airway becomes more triangular.
When saber-sheath deformity is concurrent, as is often the case, the obstruction is worsened. Atrophic rhinitis and pharyngitis may be concurrent.57 Tomograms and CT scans show typical changes, with scalloped calcification in the nodules lining the lumen of the involved segments of the airway (Figure 14-22). These nodules are within the inner border of the cartilages, which are not themselves involved. Bronchoscopy is also diagnostic (Figure 14-23). The lesions may be seen extending from the subglottic larynx to distal bronchi. In advanced cases with marked airway constriction, it may not be possible to force an adult rigid bronchoscope very far into the trachea since the process is so rigid. Biopsy is extremely difficult due to the hardness of the nodules, and it is not usually productive or necessary. Attempts to treat this condition by removal of the nodules either with bronchoscopic biopsy forceps or with laser have not been highly successful.56 The trachea may be impossible to dilate, so that per oral stent placement is not feasible. Segmental stenosis of the trachea by TPO is infrequent. Localized disease, although rare, can be resected.58,59 In 1 of our patients who had squamous cell carcinoma identified by biopsy in an obstructing localized segment of TPO, a resection gave complete relief. In 4 patients with severe and
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A B 14-22 Images of tracheopathia osteoplastica (TPO). A, Chest roentgenogram of a 48-year-old woman with TPO so severe, she was incapacitated by dyspnea. Tracheal shadow