Non malignant Tracheo-Esophageal fistula : Our experience

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1 272 Darbari et al IJTCVS Non malignant Tracheo-Esophageal fistula : Our experience Anshuman Darbari, MS, Abhishek Suryavanshi, MS, Shekhar Tandon, M. Ch., Girish Chandra, MD, Prashant Kumar Singh, MS Department of Thoracic & Cardiovascular Surgery, KGM University, Lucknow, Uttar Pradesh, India Abstract Background: Non malignant tracheo-esophageal fistula is a rare entity, which is usually post traumatic, post inflammatory or could be a delayed presentation of congenital tracheo-esophageal fistula. Patients and Methods: In this retrospective study of seven cases, we aim to document and evaluate the causes, presentation and treatment modalities. Results : All the cases underwent surgical intervention, with single stage definitive repair in four cases and two stage repair in three cases. There was no mortality, and minimal morbidity. Conclusions : Being a benign disease with fatal complications, early diagnosis and early surgical intervention is the key to successful management of non-malignant tracheo-esophageal fistula. (Ind J Thorac Cardiovas Surg 2005; 21: ) Key words: Trachea, Esophagus, Fistula Introduction Abnormal communication between trachea and esophagus, due to benign pathology, is a rare entity which can either be a late presentation of congenital tracheo-esophageal fistula (TEF) or due to acquired causes such as post traumatic, post inflammatory or secondary to any contagious diseases 1. The classical presentation is swallow-cough sequence (Ono s sign) 2, though not present in all cases, should arouse suspicion of the diagnosis. Patients and Methods Our series consist of seven patients of non-malignant tracheo-esophageal fistula that were managed at our department since April 1995 to May Out of these seven patients, four were male (57%) and three were female (43%). Age of presentation ranged from six yrs to fifty-five yrs with mean of thirty-three yrs. Duration of symptoms before presentation from time of inciting event ranged from eight hours to two months with mean Address for correspondence: Dr. Anshuman Darbari CTVS Department, KGM University Lucknow , Uttar Pradesh, India Phone No , Fax No darbarianshu@indiatimes.com IJTCVS /58 Received - 28/08/05; Review Completed - 30/09/05; Accepted - 10/10/05. period of around Seventeen days. Six patients in our series were post traumatic in origin (85.7%) and one was delayed presentation of a congenital malformation (tracheoesophageal membrane ){14.3%}. In the six patients of post traumatic origin, only one patient acquired fistula due to immediate trauma (16.7%) while origin of rest five could be assigned to post traumatic inflammation (83.3%). In the series, six patients presented with positive Ono s sign and all these patients were of thoracic fistula (85.7%) while remaining one with cervical fistula (at the level of C-7) presented with dyspnea and dysphagia. Six patients of thoracic fistula had pneumonitis of varying degrees. Our patient of cervical fistula presented within eight hours of bull horn injury neck for which tracheostomy was done immediately and ryle s tube was placed for feeding, keeping the patient nil per orally which could be the reason for absence of classical Ono s sign and pulmonary infection. Patient with congenital anomaly presented with long-standing dysphagia with recent onset of haemoptysis and positive Ono s sign. In all patients diagnosis was established and confirmed with the help of contrast x-ray of esophagus (Figs.1 & 2), esophagoscopy and bronchoscopy. Results All patients underwent surgical intervention under general anesthesia with single stage definitive repair in four patients and two stage repair in three patients. 272

2 IJTCVS Darbari et al ; 21: Tracheo esophageal fistula Fig. 1. Barium study showing a fistulous tract from esophagus to trachea in an adult patient (case no. 7) Utmost care was taken to prevent soiling of lungs by intermittent endo tracheal suction. We approached most (six out of seven) through right posterolateral thoracotomy. For cervical tracheo-esophageal fistula, neck exploration was done and repair of tubes with interposition of pedicled sternocleidomastoid muscle flap was done. For congenital tracheo-esophageal fistula, tube separation and closure of the defects was performed with interposition of pedicled intercostal. In patient work thoracic tracheoesophageal fistula (four out of seven) undergoing single stage repair, in one patient simple closure of the defects of trachea and esophagus were done, while in the remaining three patients, repair was done with interposition of pedicled intercostal. In one patient, where he presented to us with post bull horn injury, cervical fistula, initially only tracheostomy was done due to injury of larynx with ryle s tube insertion for feeding. Later on as general condition improved and laryngeal wound healed, repair of tubes with closure of tracheo-esophageal fistula was done with sternocleidomastoid flap interposition. In the second patient, where he had history of post acid ingestion, dysphagia and later positive Ono s sign, presented to us after 25 days with feeding gastrostomy, Fig. 2. Oblique view of Barium study showing a fistulous tract from esophagus to trachea in an adult patient (case no. 7). which was done at a peripheral centre as an immediate procedure to improve the nutritional status. At our center, esophagoscopy revealed non-negotiable esophageal stricture and proximal fistula. Patient underwent tracheal repair and retrosternal colonic bypass. In third patient, where patient had vomiting episode following alcohol intake and further sequence of events was not known, but patient presented to us with positive Ono s sign, after 15 days. Fistula repair by right posterolateral thoracotomy attempted but due to presence of distal esophageal stricture, diseased esophageal segment exclusion along with feeding jejunostomy was done as primary procedure, later after two months retrosternal bypass by Gavriliu reverse peristaltic stomach tube was done Patients were followed for period of 6 months to 2 years. Intra operative and postoperative mortality was nil with minimal postoperative morbidity except superficial wound infection in two patients, that responded to antibiotic therapy.[table.1]. Discussion The primitive pulmonary system develops as an out 273

3 274 Darbari et al IJTCVS Table 1. Details of all patients Age/ Sex Symptoms Time of Investigations Pathology Treatment Outcome & Signs Presentation 6yrs/ F Coin ingestion After Barium swallow Esophageal pouch Right Uneventful conservative 3 weeks and Esophago- formation with Posterolateral Management scopy tracheoesophageal thoracotomy ONO S sign positive fistula separation of tubes and closure of defects 26yrs/F Post Lime ingestion After 25 Barium swallow Esophageal Right Posterolateral Recovered but Dysphagia and days and stricture and thoracotomy morbidity +nt ONO S sign +ve Esophagoscopy, tracheoesophageal separation of tubes After 5 days at Bronchoscopy at fistula from and closure of defects peripheral center stricture to carina with interposition feeding Gastrostomy of pedicled intercostal 2nd stage Retrosternal colonic bypass 50 yrs/f Bull horn injury in After 8 Barium swallow Tracheoesophageal Tracheostomy with Uneventful neck Dyspnea with hours and fistula at cervical Ryle s tube insertion dysphagia Esophagoscopy seven (C-7) level 2 nd Stage Neck exploration separation of tubes and closure of defects with interposition of pedicled sternomastoid muscle flap 55yrs/M Denture Ingestionà After 1 Barium swallow Tracheo- esophageal Right Posterolateral Uneventful ONO S Sign + VE Months and fistula at 22 cms. thoracotomy Esophagoscopy from incisor teeth separation of tubes and closure of defects with interposition of pedicled intercostal 6 yrs./m Mango prickle After 2 Barium swallow At T3 level Right Uneventful Triangular kernel Months and Triangular Mango Posterolateral ingestion Esophagoscopy kernel stuck in the thoracotomy ONO S SIGN+VE anterior wall of the separation of tubes esophagus forming a closure of defects tracheoesophage al fistula with interposition of Pedicled intercostal 55 yrs./m Long standing After 1 Barium swallow 1 cm diameter Right Uneventful dysphagia and Months and tracheo esophageal Posterolateral regurgitation Esophagoscopy just above carina thoracotomy Recurrent without any separation of tubes Haemtemesis from adhesions and closure of one month ONO S around it defects with SIGN +VE interposition of pedicled intercostal 22 yrs./m Post alcohol intake After 15 Barium swallow Long (3cms) Right Recovered but Severe Vomiting days and proximal Posterolateral morbidity +nt ONO S SIGN +VE Esophagoscopy, tracheoesophage thoracotomy with Bronchoscopy at fistula at carina esophageal exclusion with distal esophageal structure and feeding jejunostomy 2nd Stage Retrosternal Gavriliu Gastric tube bypass 274

4 IJTCVS Darbari et al ; 21: Tracheo esophageal fistula pouching of the ventral wall of the esophagus throughout its length. Separation of the respiratory and upper gastrointestinal tracts takes place by a septum formed from two lateral grooves. The septum becomes complete caudally first and then extends upwards into the cranial direction. Any developmental arrests or failures in the completion of this septum will obviously result in tracheo-esophageal fistula. Since the esophagus increases in length at a faster rate and more than the trachea, the fistula stretches in an upward curve with the tracheal opening more cephalic than the opening into the esophagus. Tracheo-esophageal fistulas in adults are usually due to malignancy, while those due to benign pathology, are rare. Non malignant tracheo-esophageal fistula are caused by: 1. Delayed presentation of congenital tracheoesophageal fistula (all are of H-type which comprise only 2-3% of all congenital TOF) [13] or presence of tracheoesophageal membrane which gets ruptured later on in life leading to tracheo-esophageal fistula,as only such type of fistula can remain dormant till its presentation in adult where both the tubes are patent Post traumatic due to foreign body ingestion, blunt or penetrating trauma, pressure changes during vomiting, endotracheal intubation, tracheostomy 1,3,4. 3. Post inflammatory secondary to trauma, oesophagitis, caustic injury and mediastinitis. 4. Post-operative follows vagotomy for hiatus hernia, pulmonary resection and aortic aneurysm repair 1 5. Contagious diseases like tuberculosis 6, syphilis or histoplasmosis infecting lymph nodes or pulmonary infections like empyema and lung abscess 2 6. In AIDS patients, esophagitis is common, which may result in fistula formation. Most common causes are candida, mycobacterium etc 6,8. Most important investigatory tool is contrast enhanced X-ray study of esophagus, which almost in all cases demonstrate the fistula. Supplementary investigations are oesophagoscopy and bronchoscopy, which not only visualize the opening of fistula but also tell about the mucosa of both the tracts, rules out any other pathology and have some times been used as interventional tools to seal the fistula. CT scan is emerging as a very important tool not only in diagnosis of tracheo-esophageal fistula and evaluation of luminal condition, but also helps to evaluate the extraluminal condition e.g.lymph node status, to exclude any mass etc 12. A very important part of the management is pre operative preparation, which involved careful use of appropriate antibiotics to control infection and chest physiotherapy to improve the pulmonary function and measures to improve the nutritional status of patient as tracheo-esophageal fistula involves portals of two very important systems of the body thus hampers both oxygenation and nutrition of the patient 4. Few authors have suggested conservative approach in the patients on mechanical ventilation until the patient is weaned off 3. In one report of Parry et al., which describes successful treatment of a congenital bronchoesophageal fistula with endoscopic application of acetic acid and sodium hydroxide locally to bronchial and esophageal aspect of the fistula. They had suggested its use in patient, too sick to undergo thoracotomy 11, but such treatment overlooks other causes such as malignant fistula and post inflammatory fistula 7. Clinical principle in our era is that all the management decisions should, as far as possible, be evidence based. If we scrutinize the peer reviewed literature, conservative management of tracheo-esophageal fistula hardly finds a place 2,4,10. Although spontaneous healing has been reported, direct surgical repair should be the procedure of choice. In thoracic tracheo-esophageal fistula, preferred approach should be right postero lateral thoracotomy, which gives adequate exposure of esophagus, trachea and right main bronchus. In cases of tracheo-esophageal fistula above the level of clavicle, cervical approach is preferred, as in our case. Whatever the local conditions, we prefer three-layer closure of esophageal and tracheal defects, after separation of both the tubes and proper mobilization. As esophagus is devoid of serosa, reinforcement of repair site with intercostal or any other locally available muscle interposition flap is necessary. So now, it becomes three layer closure of fistula 3,15. It supports the healing and acts as barrier between these two tubes, thus preventing recurrent fistula. Esophageal bypass with transverse colon or gastric tube is done in presence of irreparably damaged esophagus or esophageal stricture. Preferred route is retrosternal approach, making this as a safer and easier one, suspecting dense adhesion in the mediastinal region. Our policy of management mainly focussed on early surgical intervention in all, with one stage repair of fistula barring those three cases where two stage repair was done for above mentioned reasons. In our series postoperative mortality was nil which can be attributed to the fact that most were intervened earlier, pulmonary infection was mild to moderate only, suggesting that non malignant tracheo-esophageal fistula have a better overall outcome. 275

5 276 Darbari et al IJTCVS References 1. Shah CP, Yeolekar ME, Pardiwala FK. Acquired tracheooesophageal fistula. J Postgrad Med 1994; 40: Gerzic Z, Rakic S, Randjelovic T. Acquired Benign Esophagorespiratory Fistula: Report of 16 Consecutive cases. Ann Thorac Surg 1990; 50: Mathisen DJ, Grillo HC, Wain JC, Hilgenberg AD. Management of Acquired Nonmalignant Tracheoesophageal Fistula. Ann Thorac Surg 1991; 52: Hilgenberg AD, Grillo HC. Acquired nonmalignant Tracheoesophageal Fistula J Thorac Cardiovasc Surg 1983; 85: Holman WL, Vaezy A, Postlethwait RW, Bridgman A. Surgical treatment of H-Type Tracheoesophageal Fistula Diagnosed in an Adult. Ann Thorac Surg. 1986; 41: Ramo OJ, Salo JA, Isolauri J, Luostarinen M, Matilla SP. Tuberculous Fistula of the Esophagus. Ann Thorac Surg 1996; 62: Ramo OJ, Salo JA,Isolauri J, Matilla SP. Congenital Bronchoesophageal Fistula in the Adult. Ann Thorac Surg 1995; 59: Temes RT, Wong RS, Davis M, Kesslar RM, Wernly JA. Esophago- Airway Fistula in AIDS. Ann Thorac Surg 1995; 60: Gudovsky LM, Koroleva NS,Biryukov YB, Chernousov AF, Perelman MI. Tracheoesophageal fistulas. Ann Thorac Surg. 1993; 55: Anderson RP, Sabiston DC Jr. Acquired bronchoesophageal fistula of benign origin. Surg gyneco obstet 1965; 121: Parry W, Juma A. Congenital bronchoesophageal fistula [letter]. Ann Thorac Surg 1991; 51: Sakamoto Y, Seki Y, Tanaka N, Nakazawa T, Nobori M.Tracheoesophageal fistula after blunt chest trauma: successful diagnosis by computer tomography. Thorac Cardiovasc Surg. 2000; 48: Haight C. Congenital tracheoesophageal fistula without esophageal atresia. J Thorac Surg.1948; 17: Postlethwait RW.Congenital Atresia and Trracheoesophageal Fistula. In: Postlethwait RW, Sealy WC, editors. Surgery of the esophagus. 1st ed. Appleton Century Crofts; pp Lee LM, Razi A. Three-layer technique to close a persistent tracheo-oesophageal fistula. Asian J Surg. 2004; 27:

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