A RARE CAUSE OF BRONCHOBILIARY FISTULA

Abstract

SESSION TITLE: Medical Student/Resident Pulmonary Manifestations of Systemic Disease 3 SESSION TYPE: Med Student/Res Case Rep Postr PRESENTED ON: 10/09/2018 01:15 PM - 02:15 PM INTRODUCTION: Bronchobiliary fistula (BBF) is a rare medical condition that presents a diagnostic dilemma. It may be congenital or acquired, with the former having a prevalence of less than 1 million. Once diagnosed its etiology is often evident, however with its incidence due to echinococcal origin being < 2 percent, patients often have multiple hospitalizations prior to diagnosis. We present a rare case of a BBF, treated successfully with a minimally invasive approach. CASE PRESENTATION: 74 year old Syrian female with prior of history of a liver cyst drained of 200 millimeters 3 years prior at another institution comes with right upper quadrant abdominal pain, nausea and vomiting. She has an abdominal magnetic resonance imaging, subsequent CT-guided fine-needle aspiration and is diagnosed with hydatid cyst, treated conservatively with albendazole. She is discharged and presents 3 days later, with productive cough of yellow/green mucus, malaise and fever. She is treated as a healthcare associated pneumonia. Sputum culture shows gram positive cocci and antibiotics are tailored from cefepime and vancomycin to cephalexin in view of MSSA. The patient improves clinically and is discharged on albendazole plus cephalexin and scheduled for elective hepatic cyst resection. She returns 8 days later for fever and productive cough. She is found to have persistent pneumonia with elevated right hemidiaphragm on chest x-ray, treated with piperacillin-tazobactam and vancomycin empirically along with albendazole for hydatid cyst. She has a laparoscopic echinococcal cyst resection with some hepatic resection to facilitate cyst removal. While on the medical floor, she develops bilioptysis and leukocytosis. A BBF is suspected and confirmed with DISIDA scan. An endoscopic retrograde cholangiopancreatography (ERCP) is done with sphincterotomy and stent placement. She continues to clinically improve and is subsequently discharged with no cough for outpatient follow-up. DISCUSSION: Acquired BBF is a rare complication of echinococcal origin. While bilioptysis is the hallmark sign of BBF it may not always be present. This poses a diagnostic dilemma for physicians and potential life threatening complications for patients. The diagnosis is made with a DISIDA. Historically treatment has been surgical, but conservative options such as embolization and stent placement have been proposed. In this case, an ERCP sphincterotomy and stent placement is successfully used. This intervention results in a decrease in the transpapillary pressure gradient, thereby facilitating biliary flow to the intestine and healing of the BBF. CONCLUSIONS: BBF of echinococcal origin has an incidence of < 2%. Bilioptysis, a clinical hallmark, maybe absent posing a clinical dilemma resulting in multiple hospitalizations. This case highlights a rare diagnosis of BBF secondary to hydatid cyst and a successful conservative option, to a once surgical problem. Reference #1: Mohammadreza Seyyedmajidi MD, Mojtaba Kiani MD, Hamid Javadi MD, Alireza Raeisi MD, Majid Assadi MD. Hepatobiliary scintigraphy with SPET in the diagnosis of bronchobiliary fistula due to a hydatid cyst. Hellenic Journal of Nuclear Medicine. 2015 May - August; 18(2):160-162. Reference #2: Guan-Qun Liao, Hao Wang, Guang-Yong Zhu, Kai-Bin Zhu, Fu-Xin Lv, Sheng Tai. Management of acquired bronchobiliary fistula: A systematic literature review of 68 cases published in 30 years. World Journal of Gastroenterology. 2011 September; 17(33): 3842-3849. Reference #3: Anton Crnjac, Vid Pivec, Arpad Ivanecz. Thoracobiliary fistulas: literature review and a case report of fistula closure with omentum majus. 2013 Radiol Oncol; 47(1): 77-85. DISCLOSURES: No relevant relationships by Sahai Donaldson, source=Web Response No relevant relationships by Killol Patel, source=Web Response No relevant relationships by Shanta Shrestha, source=Web Response