Tracheoesophageal fistula: combined surgical and endoscopic approach

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Diseases of the Esophagus (2006) 19, 36–39 © 2006 ISDE

Case report

Blackwell Publishing Asia

Tracheo-esophageal fistula: combined surgical and endoscopic approach J. P. Freire,1 S. M. Feijó,2 L. Miranda,1 F. Santos,1 H. B. Castelo1 1

Department of Surgery, Hospital de Santa Maria, Faculdade de Medicina de Lisboa and 2Department of Pulmonary Diseases, Hospital de Santa Maria, Faculdade de Medicina de Lisboa, Portugal

SUMMARY. We present a case of benign acquired tracheo-esophageal fistula caused by cuffed tracheal tube. The patient was septic and weaning from the mechanical ventilation was unlikely, so immediate repair of the tracheal and esophageal lesions was imperative. A silicone endoprosthesis was inserted in the tracheal lumen and the esophageal lesion was repaired with direct suture. After weaning from mechanical ventilation and recovery, the patient was discharged with a silicone tracheal stent. Three months later the stent was removed without complications or need for further treatment. This combined approach as never been published before, and can be a very useful alternative for the treatment of this rare but taxing complication. KEY WORDS: post entubation tracheo-esophageal fistula, tracheal stent.

keep control of the patient’s airway and allow mechanically assisted ventilation. Forty-four hours after admission an endoscopy of the respiratory tract excluded epithelial injury but weaning from the mechanical ventilation was impossible due to fatigue and low O2 saturation. By the 20th day after admission, burn lesions were grafted and already healed but the patient remained totally dependent on mechanical ventilation without any obvious explanation, and the next day, significant amounts of air were collected from the nasogastric feeding tube, in excess of 12 L for every 8 h. A diagnosis of possible tracheoesophageal fistula was considered but not objectively demonstrated. The initial plan was: establishment of spontaneous ventilation and posterior tracheobronchic endoscopy to confirm the clinical diagnosis and, eventually, surgical correction of the tracheal and esophageal lesion in accordance with what has been published and recommended in the literature.1– 4 However, 3 days later, the patient presented with a clinical picture of multiple organic failure with ARDS; impaired renal function, fever, tachycardia and shock. Inflammatory signs in the neck were present but difficult to judge due to the previous burn lesions. An esophagoscopy proved to be easier and safer to perform than the respiratory endoscopy and this confirmed the tracheo-esophageal fistula showing the tracheal tube cuff on the esophagel lumen and a vertical 4 cm tear in the anterior surface of the cervical esophagus above the level of the

INTRODUCTION Tracheo-esophageal fistula is a rare but troublesome complication of cuffed tracheal tubes. Due to its rarity, large and significant studies of this condition are not available. A few cases were reported with different clinical pictures and treatment solutions. Results and conclusions are therefore difficult to reach and there are no established guidelines for the management of this problem. Herein is described a solution combining surgical approach of the esophageal lesion and the interposition of a temporary silicone tracheal stent. We believe that, in experienced hands, this is a reproducible and reliable procedure that can be an efficient and safe alternative for this problem.

CASE REPORT A 53-year-old man was admitted to the emergency room of Santa Maria Hospital, Lisbon, with second degree burns of the face, neck and anterior surface of the chest, in a total of 20% corporeal surface. Respiratory tract burning was a strong possibility and a cuffed tracheal tube was inserted in order to

Address correspondence to: José Paulo Freire, Clínica Universitária de Cirurgia II, Piso 5, Hospital de Santa Maria, Av, Prof Egas Moniz, 1649-035 Lisboa, Portugal. Email: [email protected] 36

Tracheo-esophageal fistula 37

Fig. 3 Bronchoscopy. A silicone stent was inserted occluding the tracheal defect. Fig. 1 Esophagoscopy. Tracheal tube cuff can be seen in the right upper corner.

Fig. 2 Bronchoscopy. A 4 cm longitudinal tear in the pars membranosa can be seen allowing the tip of the nasogastric feeding tube to protrude into to the trachea.

Fig. 4 Esophagoscopy. By the 10th postoperative day the esophageal suture (left upper corner) was healed.

thoracic outlet (Fig. 1). Delayed management was no longer an option. Immediate reconstruction of the tracheal lesion was very likely destined to fail due to the need to keep the patient on mechanical assisted ventilation. We opted for the insertion of a silicone tracheal endoprosthesis to seal the tracheal defect (Figs 2, 3). We had extensive experience with these devices in benign and malign tracheal strictures. This prosthesis is usually well tolerated, sometimes for very long periods (years), with very few complications. The esophageal lesion was treated by a

direct surgical approach through a left cervical incision with direct suture of the anterior esophageal tear and cervical drainage. A nasogastric feeding tube was left in place. The patient recovered quite well. Renal and pulmonary malfunction were reverted. By the 10th postoperative day another air leak through the nasogastric feeding tube raised the suspicion of a new fistula but a new esophagoscopy ruled out this problem and showed a complete healing of the esophageal tear (Fig. 4). By the 15th postoperative day spontaneous ventilation was

38 Diseases of the Esophagus

Fig. 5 Bronchoscopy. The silicone tracheal stent was removed 3 months later with excellent functional and morphologic results.

achieved and a few days later the patient was discharged and followed-up as an outpatient. Three months later the prosthesis was removed without any signs of stenosis or tracheomalacia (Fig. 5), and the patient required no further treatment.

DISCUSSION Acquired-non-malignant tracheo-esophageal fistulas due to cuffed tracheal or tracheostomy tubes are a rare but cumbersome problem. Rarity precludes solid available experience on this subject. Even the largest report does not exceed 38 cases,2 and overall, in the last 42 years, only 172 cases have been reported. They are very different in their clinical picture, in the solutions adopted, and naturally, in the results. Mortality is frequent, from 3% to 80% according to clinical presentation and the solutions adopted (surgical vs. conservative).5,6 Morbidity is also substantial, and a large number of patients require more than one procedure. Generally, delayed reconstruction of the tracheal and esophageal lesion is recommended, after establishment of spontaneous ventilation, mainly because surgical repair of tracheal lesions under positive pressure ventilation is usually not successful due to an increased risk of anastomotic dehiscence and restenosis. However, as happened with our patient, this is not always possible. Major surgical procedures are normally suggested, namely tracheal sleeve resection and anastomosis and esophageal repair, with or without tissue interpositions (muscle flaps).7–9 The authors who used surgical procedures which included esophageal resection, exclusion and bypass, recognized the inherent morbidity and mortality that is associated with them.10,11 Double stenting is not recommended due to the

high potential risk of fistula enhancement and the unlikelihood of tissue healing between prostheses.12 The proposed solution came from 12 years’ experience with interventional bronchoscopy with silicone stents in the treatment of benign and malign tracheo-bronchial stenosis and tracheo-esophageal fistulas of multiple etiologies. This experience, together with surgical expertise on esophageal surgery is necessary to deal with these situations and should be available on every tertiary referral medical center to which such patients may be sent. This combined and complementary approach allows closure of the tracheal lesion although avoiding major tracheal procedures, and also allows simple and easy suture of the esophageal lesion. These silicone stents (Dumon or Hood stents) are easy to insert and remove at any time. Metallic stents, expansible or auto-expansible should be avoided because they are prone to complications and difficult to remove. Esophageal lesions located at the thoracic outlet, where surgical approach can be cumbersome, can be dealt with via double esophageal stapling, above and below the perforation, and with a feeding jejunostomy. Normally, as with our experience and also in the literature,13–15 after 2–3 weeks, the esophageal lumen spontaneously reopens and by that time the wall defect is healed. This, we consider, is an effective, simple and safe approach for the rare but distressing problem of iatrogenic tracheo-esophageal fistulas. References 1 Grillo H C. Surgical treatment of post-intubation lesions of the trachea. Acta Chir Belg 1977; 76: 361–9. 2 Mathisen D J, Grillo H C, Wain J C, Hilgenberg A D. Management of acquired nonmalignant tracheoesophageal fistula. Ann Thorac Surg 1991; 52: 759–65. 3 Reed M F, Mathisen D J. Tracheoesophageal fistula. Chest Surg Clin N Am 2003; 13: 271–89. 4 Hilgenberg A D, Grillo H C. Acquired nonmalignant tracheoesophageal fistula. J Thorac Cardiovasc Surg 1983; 85: 492–8. 5 Macchiarini P, Verhoye J P, Chapelier A, Fadel E, Dartevelle P. Evaluation and outcome of different surgical techniques for postintubation tracheoesophageal fistulas. J Thorac Cardiovasc Surg 2000; 119: 268–76. 6 Marzelle J, Dartevelle P, Khalife J, Rojas-Miranda A, Chapelier A, Levasseur P. Surgical management of acquired post-intubation tracheo-oesophageal fistulas: 27 patients. Eur J Cardiothorac Surg 1989; 3: 499–502; discussion 502–3. 7 Oliaro A, Rena O, Papalia E et al. Surgical management of acquired non-malignant tracheo-esophageal fistulas. Cardiovasc Surg (Torino) 2001; 42: 257–60. 8 Wolf M, Yellin A, Talmi Y P, Segal E, Faibel M, Kronenberg J. Acquired tracheoesophageal fistula in critically ill patients. Ann Otol Rhinol Laryngol 2000; 109: 731–5. 9 Villalba-Caloca J, Tellez-Becerra J L, Morales-Gomez J, Molina-Barrera E A, Lopez-Flores D. Surgical treatment of non-malignant tracheoesophageal fistula. Gac Med Mex 1998; 134: 397– 405. 10 Baisi A, Bonavina L, Narne S, Peracchia A. Benign tracheoesophageal fistula: results of surgical therapy. Dis Esophagus 1999; 12: 209–11. 11 Cherveniakov A, Tzekov C, Grigorov G E, Cherveniakov P. Acquired benign esophago-airway fistulas. Eur J Cardiothorac Surg 1996; 10: 713–6.

Tracheo-esophageal fistula 39 12 Bugge-Asperheim B, Birkeland S, Storen G. Tracheooesophageal fistula caused by cuffed tracheal tubes. Scand J Thorac Cardiovasc Surg 1981; 15: 315–9. 13 Laddin D A, Dunnington C W, Kirby C K. Stappled esophageal exclusion in acute esophageal rupture: a new technique. Contemp Surg 1989; 35: 45.

14 Bardini R, Bonavina L, Pavanello M et al. Temporary double exclusion of the perforated esophagus using absordable staples. Ann Thorac Surg 1992; 54: 1165. 15 Venki P, Rumisek J D, Chang C C. Spontaneous recanalization of the esophagus after exclusion using nonabsorbable staples. Ann Thorac Surg 1995; 59: 1214.

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