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 ::  Introduction
 ::  Case Report
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CASE REPORT
Year : 2010  |  Volume : 56  |  Issue : 4  |  Page : 284-286

Endoscopic occlusion of idiopathic benign esophago-bronchial fistula


1 Department of Gastroenterology, Narayana Medical College, Nellore, India
2 Department of Internal Medicine, Creighton University, Medical Center, Nebraska, USA
3 Department of Surgery, Narayana Medical College, Nellore, India

Date of Submission16-Jan-2010
Date of Decision22-Apr-2010
Date of Acceptance16-Jun-2010
Date of Web Publication7-Oct-2010

Correspondence Address:
R K Yellapu
Department of Gastroenterology, Narayana Medical College, Nellore
India
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DOI: 10.4103/0022-3859.70941

PMID: 20935400

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 :: Abstract 

We report a 35-year-old male with recurrent respiratory infections and cough associated with ingestion of food for 15 years. He was diagnosed to have benign esophago-bronchial fistula and proximal jejunal stricture secondary to a tumor, which was surgically resected. In view of recent surgery, endoscopic closure of the fistula was attempted initially with an endoclip resulting in partial symptomatic relief. The fistula was later completely occluded endoscopically with cyanoacrylate glue. A barium swallow at eight-months follow-up revealed no evidence of esophago-bronchial communication. A high index of suspicion is required in the diagnosis of this rare entity in adults with recurrent respiratory infections of obscure etiology. Surgical resection is the standard treatment. Here we report a rare case of idiopathic benign esophago-bronchial fistula in an adult, treated with endoscopic approach.


Keywords: Endoclip, endoscopic glue occlusion, idiopathic esophago-bronchial fistula


How to cite this article:
Yellapu R K, Gorthi J R, Kiranmayi Y, Sireesh I. Endoscopic occlusion of idiopathic benign esophago-bronchial fistula. J Postgrad Med 2010;56:284-6

How to cite this URL:
Yellapu R K, Gorthi J R, Kiranmayi Y, Sireesh I. Endoscopic occlusion of idiopathic benign esophago-bronchial fistula. J Postgrad Med [serial online] 2010 [cited 2014 Dec 22];56:284-6. Available from: http://www.jpgmonline.com/text.asp?2010/56/4/284/70941



 :: Introduction Top


Broncho-esophageal fistulae in adults are mostly acquired in nature. Surgical resection is the treatment modality of choice. Here we report a case of idiopathic benign esophago-bronchial fistula in an adult, treated by endoscopic approach.


 :: Case Report Top


A 35-year-old male presented with worsening cough and bilious vomiting for one month. Past history was significant for recurrent respiratory infections and cough associated with ingestion of food for 15 years. Physical examination was unremarkable. Complete blood count, blood chemistry, chest and abdominal radiographs were normal. Barium swallow showed a distal esophago-bronchial fistula [Figure 1]a and b. Esophago-gastroduodenoscopy (EGD) revealed lower third esophageal diverticulum [Figure 2]a, with reflux esophagitis and significant bilious gastric residue. He had a proximal jejunal stricture, secondary to tumor which was resected with wide margins. Although bilious vomiting improved postoperatively, he had persistent cough with ingestion of food or drink (Ohno's sign). In the absence any evidence of tuberculosis, histoplasmosis, malignancy, trauma or chest wall surgery and long duration of symptoms , idiopathic benign esophago-bronchial fistula was diagnosed. Endoscopic closure with triclip was initially attempted, with partial relief [Figure 2]b. EGD was repeated after a week for repeat therapeutic interventions. A 10-French endoscopic retrograde cholangiopancreatography (ERCP) cannula (Wilson Cook Medical, Winston Salem, USA) was advanced through the endoscope and positioned into the fistula [Figure 2]c, with the cannula tip exiting a few centimeters from the tip of the endoscope, 2 ml of N-butyl-2-cyanoacrylate glue (Gesika, Reevax, Hyderabad, India) diluted in lipoidol was applied through the cannula and endoscope was slowly withdrawn without retreating the cannula to prevent occlusion of scope channel. EGD showed the fibrin glue plug at the fistula opening [Figure 2]d. Barium swallow after the procedure revealed complete occlusion of the fistula. The procedure was done with fluoroscopic guidance to visually monitor airway during glue injection. The patient had symptomatic improvement with no episodes of cough associated with ingestion of food and was discharged three days after the procedure. He was asymptomatic at 12-month follow-up and a barium swallow showed no evidence of fistula [Figure 3].
Figure 1 :(a, b) Barium esophagogram showing contrast extravasation from distal third of the esophagus through a fistulous tract into the left lower lobe posterior segmental bronchus

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Figure 2 :(a) Wide-necked esophageal diverticulum at 34 cm from incisors. (b) Endoclip closing the fistula. (c) ERCP cannula positioned in the fistula. (d) Cyanoacrylate glue occluding the fistula

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Figure 3 :Barium esophagogram showing no evidence of broncho-esophageal fistula after endoscopic glue occlusion

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 :: Discussion Top


Benign esophago-bronchial fistulas are rare and, owing to the combination of their rarity and the nonspecific nature of the symptoms, they remain undiagnosed for a long time. Common causes of benign esophago-bronchial fistulas are prior esophageal surgery, infections like tuberculosis and histoplasmosis, lye ingetion and congenital malformation. Sometimes, despite extensive investigations, the cause is not apparent (Idiopathic).

This anomaly commonly results in episodes of cough associated with ingestion of food (Ohno's sign) and recurrent pulmonary infections. Diagnosis is established by barium swallow, bronchoscopy, and endoscopy or computed tomography of chest. Surgical intervention in the form of fistulectomy and closure still remains the standard of care. [1],[2] Alternative modes of management suggested earlier include: conservative management using Celestin tube to exclude esophageal secretions from the fistula and thus mitigate pulmonary contamination, [3] or painting the fistula site with 10% sodium hydroxide solution followed by a 30% acetic acid solution via bronchoscope. [4]

Diverse endoscopic therapies have been reported to treat benign esophago-bronchial fistulas. [5],[6],[7],[8] These include various occlusion therapies, endoclips, covered metallic stents and laser or combined endoscopic therapies. [9],[10] Different endoscopic occlusion therapies tried include: fibrin tissue, fibrin glue, vicryl plug and glue and a new liquid polymer sealant "Onyx". [8] Endoscopic sealing treatment achieves a very high success rate, without significant complications and at a lower cost. It could reduce hospital stay and avoid surgery in high-risk patients.

The tissue glue, N-butyl-2-cyanoacrylate is commonly used as topical tissue adhesive and to stop oozing from tissues. In gastroenterology practice it is used for obliteration of gastric varices, fistula and certain leaks. [11],[12] N-butyl-2-cyanoacrylate is a watery solution that polymerizes and hardens instantaneously, within 4 seconds. There have been reports of needles becoming stuck in the N-butyl- 2-cyanoacrylate and subsequent tearing of the vessel on withdrawal of the needle. To prevent this, it is necessary to dilute it with oily contrast agent Lipoidol. Patients with allergies to iodine should not receive this therapy because Lipoidol is an iodized oil emulsion. This dilution also allows for visualization when using fluoroscopy to localize the injection. One of the problems described with the use of glue is accidental adhesion to the scope lens and damage to the endoscope; however, immediate removal with Acetone or Dimethyl Formide mitigates this problem.

In our patient, no apparent cause of esophago-bronchial fistula was found. In view of recent abdominal surgery in this patient, we attempted the closure of fistula using an endoclip initially; however, there was only partial symptomatic relief. Later we applied cyanoacrylate glue into the fistula which formed a tight sealant, occluding the fistulous tract. He remained asymptomatic at last follow-up, (12 months after procedure). There are a few similar reports of successful glue occlusion in patients with benign esophago-bronchial fistula in the literature, however, without long-term follow-up. [13],[14] Endoscopic glue therapy is not effective in post-infectious fistula.

Therapeutic endoscopic glue application is also reported in closing persistent biliary leaks post cholecystectomy, [15] postoperative colorectal fistula, [16] entero-cutaneous fistula, and pancreato-cutaneous fistula [17] [Table 1]. Non-gastrointestinal utilities are reported in plastic surgery, for example in maintaining brow position in endoscopic browplasty. [18] Glue has also been reported to be effective in closure of vesico-vaginal fistula [19] and broncho-pleural fistula. [20]
Table 1 :Applications of endoscopic glue


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Endoscopic glue occlusion is an alternative to surgery, especially in high-risk patients with complex needs. Further, it can be done on an outpatient basis, is less expensive and can be repeated if necessary. However, long-term success needs to be determined.

 
 :: References Top

1.Ramo OJ, Salo JA, Mattila SP. Congenital bronchoesophageal fistula in the adult. Ann Thorac Surg 1995;59:887-90.  Back to cited text no. 1      
2.Su L, Wei XQ, Zhi XY, Xu QS, Ma T. Congenital bronchoesophageal fistula in the adult: A case report. World J Gastroenterol 2007;13:3776-7.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]  
3.Laforet EG. Treatment of esophago-bronchial fistula. J Am Med Assoc 1978;239:2388.  Back to cited text no. 3      
4.Parry GW, Juma A, Dussek JE. Bronchoesophageal fistula treated effectively without surgical resection. Thorax 1993;48:189-90.   Back to cited text no. 4  [PUBMED]  [FULLTEXT]  
5.Truong S, Bohm G, Klinge U, Stumpf M, Schumpelick V. Results after endoscopic treatment of postoperative upper gastrointestinal fistulas and leaks using combined vicryl plug and fibrin glue. Surg Endosc 2004;18:1105-8.  Back to cited text no. 5      
6.Cellier C, Landi B, Faye A, Wind P, Frileux P, Cugnenc PH, et al. Upper gastrointestinal tract fistula: Endoscopic obliteration with fibrin sealant. Gastrointest Endosc 1996;44:731-3.  Back to cited text no. 6  [PUBMED]    
7.De Giacomo T, Francioni F, Venuta F, Rendina EA, Ricci C. Benign esophageal-respiratory fistulae: The surgical treatment and results of 10 cases. Minerva Chir 1993;48:311-6.  Back to cited text no. 7  [PUBMED]    
8.Rieder F, Hamer O, Gelbmann C, Schφlmerich J, Gross V, Feuerbach S, et al. Crohn's disease of the esophagus: Treatment of an esophagobronchial fistula with the novel liquid embolic "onyx". Z Gastroenterol 2006;44:599-602.  Back to cited text no. 8      
9.Murdock A, Morrehead RJ, Tham TC. Closure of benign bronchoesophageal fistula with endoclips. Gastrointest Endosc 2005;62:635-8.  Back to cited text no. 9      
10.Luca GD, Griffo S, Monaco M, Fraioli G, Tommaso LD, Stassano P. Combined endoscopic approach in treatment of benign broncho- esophageal fistula. Thorax 2008;63:1024-5.  Back to cited text no. 10      
11.Bozkurt M, Kulachi Y, Zor F, Kapi E. N-butyl-2-cynoacrylate usage for controlling oozing bleeding during surgery in case of neurofibromatosis type 1. Eur J Plast Surg 2008;31:193-5.  Back to cited text no. 11      
12.McAvoy NC, Hayes PC. Endoscopic cyanoacrylate therapy for gastric variceal bleeding. Nat Rev Gastroenterol Hepatol 2010;7:190-1.  Back to cited text no. 12  [PUBMED]  [FULLTEXT]  
13.Ogunmola N, Wyllie R, McDowell K, Kay M, Mahajan L. Endoscopic closure of Esophagobronchial fistula with fibrin glue. J Pediatr Gastroenterol Nutr 2004;38:539-41.  Back to cited text no. 13  [PUBMED]  [FULLTEXT]  
14.Jones C, Laurence BH, Faulkner KW, Cullingford GL. Closure of benign broncho-oesophageal fistula by endoscopic injection of bovine collagen, cyanoacrylate glue and gelfoam. Aust N Z J Surg 1996;66:53-5.  Back to cited text no. 14  [PUBMED]    
15.Wright G, Jairath V, Reynolds M, Shidrawi RG. Endoscopic glue injection for persistent biliary leakage. Gastrointest Endosc 2009;70:1279-80.  Back to cited text no. 15  [PUBMED]  [FULLTEXT]  
16.Rotondano G, Viola M, Orsini L, Cipolletta F, Bianco MA, Garofano ML, et al. Uncommon cause of early postoperative colonic fistula successfully treated with endoscopic acrylate glue injection. Gastrointest Endosc 2008;67:183-5.  Back to cited text no. 16  [PUBMED]  [FULLTEXT]  
17.Jang JW, Park DH, Moon SH, Lee SS, Seo DW, Lee SK, et al. Endoscopic fibrin glue injection for closure of pancreatocutaneous fistula following transgastric endoscopic necrosectomy. World J Gastroenterol 2008;14:6093-5.  Back to cited text no. 17  [PUBMED]  [FULLTEXT]  
18.Sidle DM, Loos BM, Ramirez AL, Kabaker SS, Mass CS. Use of Bioglue surgical adhesive for brow fixation in endoscopic browplasty. Arch Facial Plast Surg 2005;7:393-7.  Back to cited text no. 18      
19.D'Arcy FT, Jaffry S. The treatment of vesicovaginal fistula by endoscopic injection of fibrin glue. Surgeon 2010;8:174-6.  Back to cited text no. 19  [PUBMED]  [FULLTEXT]  
20.Scappaticci E, Ardissone F, Ruffini E, Baldi S, Revello F, Coni F. Postoperative bronchopleural fistula: Endoscopic closure in 12 patients. Ann Thorac Surg 2000;69:1629-30.  Back to cited text no. 20  [PUBMED]  [FULLTEXT]  


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1]

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