Journal of Postgraduate Medicine Medknow Publications and Staff Society of Seth GS Medical College and KEM Hospital, Mumbai, India ISSN: 0022-3859 EISSN: 0972-2823 Vol. 56, Num. 4, 2010, pp. 284-286

Journal of Postgraduate Medicine, Vol. 56, No. 4, October-December, 2010, pp. 284-286

Case Report

Endoscopic occlusion of idiopathic benign esophago-bronchial fistula

RK Yellapu¹, JR Gorthi², Y Kiranmayi¹, I Sireesh³

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

Correspondence Address: R K Yellapu, Department of Gastroenterology, Narayana Medical College, Nellore, India, radhayellapu@gmail.com

Date of Submission: 16-Jan-2010
Date of Decision: 22-Apr-2010
Date of Acceptance: 16-Jun-2010

Code Number: jp10083

PMID: 20935400

DOI: 10.4103/0022-3859.70941

Abstract

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

Introduction

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

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].

Discussion

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]

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

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