Primary carcinoma of the trachea with sleeve resection and anastomosis of the circumferential defect A case report W. M. Swenson, M.D., R. J. Jensik, M.D., F. Milloy, M.D., T. L. Ashcroft,
M.D.,
and P. H. Holinger, M.D., Chicago, III.
-Lrimary carcinoma of the trachea is un common, there being about 200 cases re ported in the literature. 10 ' "· 2 4 · 25>26 When trachéal carcinoma occurs, it poses several surgical problems to which there can be various solutions. In only a very few in stances have previous investigators reported repairing circumferential defects of the tra chea by direct anastomosis.4' 21 In some instances, prostheses of different types have been utilized for trachéal replace ment.9· 19 A series of cases has recently ap peared in which lesions of the trachea and carina were removed while the patient was supported on one lung anesthesia.15 Cardiopulmonary bypass has also been employed.1 This case illustrates the manner in which a 3.7 cm. defect in the trachea was repaired by an end-to-end anastomosis without the use of cardiopulmonary bypass after a tra chea! tumor was removed. Case report The patient (E. A.) was a 58-year-old white woman who entered the hospital with the chief From the Departments of Surgery, Presbyterian-St. Luke's Hospital, and University of Illinois College of Medi cine, Chicago, 111. Received for publication June 3, 1965.
complaint of difficulty in breathing for the pre vious 4 years. She had stopped smoking cigarettes 4 years previously because she had begun to have intermittent episodes of exertional dyspnea related to it. Exertional dyspnea progressed until finally she had to rest after walking one hundred yards or after climbing fifteen steps. In the past 3 months, her family had noticed that she was wheezing almost continuously and they insisted that she seek help for her "asthma." She coughed occasionally in the morning and at these times would produce about one-half teaspoonful of white, thin, watery sputum. She had no chest pain, no hemoptysis, no weight loss, and no other sys temic complaints except chronic constipation. She took no medications. She slept on one pillow at night. On physical examination she was a normally developed, well-nourished white woman in ap parent good health. The pulse was 88 and regu lar, the blood pressure was 130/80 mm. Hg, the temperature was 98.6° F., and the respirations were 20 per minute. Pertinent physical findings were limited to the chest. The trachea was of nor mal size in the midline. Directly over the trachea a loud wheeze was heard with inspiration and ex piration. The heart and lungs were normal on physical examination. The hemogram and urinalysis were within nor mal limits. Numerous sputum specimens were negative when examined for malignant cells. A sputum culture produced a few colonies of strep tococci, Neisseria, and Micrococci. The chest roentgenogram showed a lesion just above the carina (Fig. 1). This lesion was better 545
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Fig. 3. The tumor as seen through a bronchoscope.
Fig. 1. Preoperative roentgenogram of the patient.
Fig. 2. A laminographic cut in the anteroposterior plane which shows the mass protruding from the right wall of the trachea into the lumen just above the carina.
defined by laminography (Fig. 2). At bronchoscopy a lobulated vascular tumor was seen pro truding into the lumen of the distal trachea caus ing almost complete occlusion (Fig. 3). This lesion was biopsied and was found to be a locally in vasive carcinoma of adnexal gland origin. Operation. An operation was performed on June 25, 1964. Anesthesia was begun by means of a 43 cm. endotracheal tube which was inserted to a level 6 cm. below the vocal cords. The pa tient was then placed on her left side, and the right thorax was opened. The trachea was ex posed. The lesion was found to protrude outside the trachéal confines as well as into the lumen. There was no invasion of adjacent structures by the tumor. An incision was next made in the lower trachea just distal to the lesion. The tumor mass was then pushed laterally and the endo tracheal tube was guided past it into the left main bronchus. The patient's respiration was then main tained by the left lung. A sleeve of trachea com posed of the entire lumen, three trachéal rings, associated posterior wall, and the tumor, was re moved. The resulting circumferential defect mea sured 3.7 cm. in length (Fig. 4). The severed ends of the trachea were approxi mated over the endotracheal tube by interrupted 3-0 silk sutures. The chest was closed. The pa tient tolerated the entire procedure with no diffi culty. Postoperative course. Although she coughed fairly well and was able to clear the trachéal secre tions, one bronchoscopy was performed on the third postoperative day. She has been bronchoscoped at intervals since the procedure and has showed good healing of the suture line. Now, 9 months later, she has returned to normal activity, free of her exertional dyspnea and wheeze.
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Fig. 4. The resected specimen. Left: In the side view, trachea wall is indicated by the arrow. The intraluminal portion of tumor protrudes to the left, the extratracheal portion to the right. Adjacent is the grayish cut surface of tumor. Right: The intraluminal aspect with tumor mass on the right. Upper and lower trachéal rings had previously been removed for histologie study. Pathology. The gross lesion measured 1.9 cm. in diameter. Microscopically, it was described as having irregular nests of tumor cells reminiscent of a mixed salivary gland tumor with occasional small nests of neoplastic cells such as are seen in a cylindroma. The final diagnosis of the tumor was adenocarcinoma of adnexal gland origin, clinically from the trachea, completely excised (Fig. 5).
Discussion In a recent review article, Salm26 found that there are about 200 reported cases of primary carcinoma of the trachea. He noted also that the incidence of the lesion is less than one in ten thousand necropsies and that the occurrence has remained fairly con stant in this century, in contrast with the marked increase in bronchogenic car cinoma. Although histologie classification of these carcinomas was somewhat confused in ear lier terminology because of a multitude of classifications, the epidermoid type seems most prevalent. The less common adenocarcinomas generally fall into two groups. They may have more invasive character istics, similar to the epidermoid type, or they may be the less malignant adenocarcinomas. 22 · 26 This latter tumor is the type found in the case herein described. This particular tumor occurs equally in the sexes, metastasizes rarely, and carries a good prog nosis. Of 11 patients with these low-grade malignancies recorded since 1947, 5 have died: 1 postoperatively, 3 within 12 months, and 1 after 4 years. Six patients were alive
Fig. 5. Microscopic appearance of the adenocar cinoma of adnexal gland origin with trachéal epithelium above, and nests of tumor cells below.
and well after periods ranging from 11 months to 5 years.26 Radiation therapy in trachea! carcinoma is considered of little value by some24 and advocated for routine use by others.10 Con ventional therapy, radon seed implanta tion,23· 28 radioactive cobalt, and even a four millivolt linear accelerator have been em-
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ployed with an occasional 5-year survivor.11 Because of the nature of the tumor in this patient and because the margins of the speci men at the point of resection were free of tumor, radiation therapy was not used. The surgical management of trachéal tu mors has varied. In 1 case reported by Clagett and his associates,8 the patient had been successfully treated for 6 years by re peated local excision of the lesion performed bronchoscopically. Much investigative work has sought a tra chéal substitute for use following sleeve re section of the trachea. Autogenous free tra chéal transplants were studied in dogs and the characteristics of regenerated respiratory epithelium reported. 5 · 6 Harrington and his colleagues16 used Marlex mesh to replace circumferential defects and reported epithelization over the mesh within 3 months. Ex perimental homografts have been used in animals to replace trachéal segments but have not proved satisfactory.14-1S Prosthetic tubes of vitallium and stainless steel have been used by some to replace tra chéal segments,13 whereas Clagett7 and Craig9 and their co-workers employed poly ethylene. Disadvantages were dislodgement of the tubes,7 stenosis of the trachea,16 and regeneration of the trachéal epithelium along the outside of the tubes with a resultant lack of inner cleansing action. Kramish and Morfit19 used Teflon prostheses in humans to bridge defects of 4 inches in length and 6 inches in length. Both of these patients had tracheostomies to fa cilitate the clearing of secretions. One died 9 months after surgery and showed evidence of fracture of the prosthesis beneath the tracheostomy opening where the tracheostomy tube had caused erosion. The distal anastomosis was intact. Ellis and associates12 employed a 6 cm. tubular trachéal substitute of heavy Marlex mesh in 1 patient in conjunction with a per manent tracheostomy and resection of a ma lignancy. The prosthesis had functioned for 18 months at the time of their report. Craig and associates9 performed an unsuccessful resection of the anterior half of the trachea
for epidermoid carcinoma and inserted a polyethylene tube of appropriate caliber into the defect. Belsey2 in 1950 stated that window resec tion of the trachea is likely to result in local recurrence and set forth four criteria for trachéal resection and reconstruction. These criteria are: Lateral rigidity, longitudinal elasticity and flexibility, an adequate and air-tight lumen, and an uninterrupted lining of ciliated epithelium. These criteria seem best satisfied by trachéal sleeve resection with primary anastomosis. Mathey and Oustrieres21 reported the suc cessful end-to-end anastomosis of the left "stem" bronchus after accidental division. They mentioned the technique of bringing the suture line beneath the mediastirial pleura and discussed other methods of re pair. In 1954, MacManus and McCormick20 reported a successful 3.5 cm. sleeve resec tion of the trachea. Fascia lata was used to cover a small air leak. Other circumferential resections have also been successful.3-15,2T The question frequently arises of how much tension can safely exist at the point of trachéal anastomosis. A series of animal experiments was carried out to find the an swer.4 Defects were created, varying from 5 to 15 cm. long, and the tension required to approximate the trachéal ends was mea sured before anastomosis. It was found that success was rarely achieved when suture line tensions exceeded 2,200 grams. Cardiopulmonary bypass as a necessary adjunct in these cases has been advocated. In the case of pulmonary bypass, described by Adkins, 1 a cylindroma was successfully removed from the trachea in an almost iden tical location as the tumor herein reported. It was of similar magnitude but was located on the left trachea! wall at the carina. Cardiopulmonary bypass was available to us had it been required. It was found, how ever, that a long endotracheal tube could be passed beyond the tumor once the tra chea had been opened and the tumor pushed laterally from its original occluding position. Information obtained at bronchoscopy was utilized in performing the intubation so as
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not to impinge on the tumor prior t o thoracotomy and thus avoid possible asphyxia from hemorrhage, tumor dislodgement, o r
complete occlusion of the trachea. Summary A case of primary carcinoma of the tra chea removed by sleeve resection, which re sulted in a 3.7 cm. circumferential trachea! defect, is presented. The trachea was re paired with an end-to-end anastomosis em ploying left lung anesthesia. REFERENCES 1 Adkins, P. C , and Izawa, E. M.: Resection of Trachéal Cylindroma Using Cardiopulmonary Bypass, Arch. Surg. 88: 405-409, 1964. 2 Belsey, R.: Resection and Reconstruction of the Intrathoracic Trachea, Brit. J. Surg. 38: 200-205, 1950. 3 Björk, V. O., Carlens, E., and Crafoord, C : The Open Closure of the Bronchus and the Resection of the Carina and of the Trachea! Wall, J. THORACIC SURG. 23: 419, 1952.
4 Cantrell, J. R., and Folse, J. R.: Repair of Circumferential Defects of the Trachea by Di rect Anastomoses: Experimental Evaluation, J. THORACIC & CARDIOVAS. SURG. 42: 589-598,
1961. 5 Carter, M. G., and Strieder, J. W.: Resection of the Trachea and Bronchi: An Experimental Study, J. THORACIC SURG. 20: 613-627, 1950.
6 Correll, N. O., and Beattie, E. J., Jr.: The Characteristics of Regeneration of Respiratory Epithelium, Surg., Gynec. & Obst. 103: 209211, 1956. 7 Clagett, O. T., Grindlay, J. H., and Moersch, H. J.: Resection of the Trachea: An Experi mental Study and Report of a Case, Arch. Surg. 57: 253, 1958. 8 Clagett, O. T., Moersch, H. J., and Grindlay, J. H.: Intrathoracic Trachéal Tumors: Develop ment of Surgical Techniques for Their Re moval, Tr. Am. S. A. 70: 224-236, 1952. 9 Craig, R. L., Holmes, G. W., and Shabart, E. J.: Trachéal Resection and Replacement With a Prosthesis, J. THORACIC SURG. 25: 384-396,
1953. 10 Culp, O. S.: Primary Carcinoma of the Tra chea, J. THORACIC SURG. 7: 471-487, 1938.
11 Dalbey, J. E., and McNab Jones, R. F.: Pri mary Malignant Growths of the Trachea, Acta Otolaryng. 53: 12-20, 1951. 12 Ellis, P. R., Jr., Harrington, O. B., Beall, A. C , Jr., and De Bakey, M. E.: The Use of Heavy Marlex Mesh for Trachea! Reconstruc
tion Following Resection for Malignancy, J. THORACIC & CARDIOVAS. SURG. 44: 520-527,
1962. 13 Gebauer, P. W.: Further Experience With Dermal Grafts for Healed Tuberculosis Stenosis of the Bronchi and Trachea, J. THORACIC SURG. 20: 628-651, 1950.
14 Gebauer, P. W.: Reconstructive Tracheobronchial Surgery, S. Clin. North America 36: 893, 1956. 15 Grillo, H. C , Bendixen, H. H., and Gephart, T.: Resection of the Carina and Lower Trachea, Ann. Surg. 158: 889-893, 1963. 16 Harrington, O. B., Beall, A. C , Jr., Morris, G. C , Jr., and Usher, F. C : Circumferential Replacement of the Trachea With Marlex Mesh, Am. Surgeon 28: 217, 1962. 17 Holinger, P. H., Novak, F. J., and Johnston, K. C : Tumors of the Trachea, Laryngoscope 60: 1086, 1950. 18 Jackson, T. L„ O'Brien, E. J., Tuttle, W., and Meyer, J.: The Experimental Use of Homogeneous Trachéal Transplants in the Restoration of Continuity of the Tracheobronchial Tree, J. THORACIC SURG. 20: 598-611, 1950. 19 Kramish, D., and Morfit, H. M.: The Use of a Teflon Prosthesis to Bridge Complete Sleeve Defects in the Human Trachea, Am. J. Surg. 106: 704-708, 1963. 20 MacManus, J. E., and McCormick, R.: Re section and Anastomosis of the Intrathoracic Trachea for Primary Neoplasms, Ann. Surg. 139: 350-354, 1954. 21 Mathey, J., and Oustrieres, G.: End-to-End Bronchial Anastomosis After Accidental Division of the Stem Bronchus, Thorax 6: 71-74, 1951. 22 McDonald, J. R.: Pathologic Aspects, Proc. Mayo Clin. 21: 416-426, 1946. 23 Moersch, H. J., Clagett, O. T., and Ellis, F. J., Jr.: Tumors of the Trachea, Med. Clin. North America 38: 1091-1096, 1954. 24 Pfeifer, W. M., Miller, D., and Robinson, H.: Epidermoid Carcinoma of the Trachea, Wis consin M. J. 57: 345-349, 1958. 25 Ranke, E. J., Presley, S. S., and Holinger, P. H.: Tracheogenic Carcinoma, J. A. M. A. 182: 519, 1962. 26 Salm, R.: Carcinoma of the Trachea, Brit. J. Dis. Chest 58: 61-71 and 92-94, 1964. 27 Weisel, W., Claudon, D. B., and Darin, J. C : Trachéal Adenoma in Juxtaposition With a Mediastinal Bronchogenic Cyst, J. THORACIC SURG. 37: 687, 1959.
28 Winston, P.: Carcinoma of the Trachea Treated by Radon Seed Implantation, J. Laryng. & Otol. 72: 496-499, 1958.