Ann Thorac Surg 2000;70:1392-1394
© 2000 The Society of Thoracic Surgeons
Case report
Bronchobiliary fistula: principles of management
Heidi K. Chua, MDa,
Mark S. Allen, MDa,
Claude Deschamps, MDa,
Daniel L. Miller, MDa,
Peter C. Pairolero, MDa
a Division of General Thoracic Surgery, Mayo Clinic and Mayo Foundation, Rochester, Minnesota, USA
Address reprint requests to Dr Allen, Department of Surgery, Mayo Clinic, 200 First St SW, Rochester, MN 55905
e-mail: allen.mark{at}mayo.edu
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Abstract
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Bronchobiliary fistula is an uncommon entity. Recently, we encountered 2 patients with this problem. Both were treated successfully with resection of the involved pulmonary tissue and interposition of viable tissue between the lung and the fistulous tract. This approach, although invasive, provided a rapid resolution of the patients’ problem.
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Introduction
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Bronchobiliary fistula is a relatively uncommon entity. Most literature regarding bronchobiliary fistulae are reports of complications of hydatid cyst disease or congenital bronchobiliary fistula. Other causes of bronchobiliary fistulae are traumatic, iatrogenic, and postoperative complications of hepatobiliary surgery. We recently encountered 2 patients who developed a bronchobiliary fistula after hepatobiliary surgery.
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Case reports
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Patient 1
A 47-year-old white female underwent a cholecystectomy for gangrenous cholecystitis. She had an uneventful recovery until she presented with right upper quadrant pain and fever 3 weeks postoperatively. A subdiaphragmatic abscess was diagnosed by computed tomography (CT), and CT-directed percutaneous drainage was instituted.
Four months later, she presented with pleuritic chest pain and a cough productive of yellow sputum. Chest CT demonstrated consolidation of the lateral basal segment of the right lower lobe (Fig 1). Abdominal CT demonstrated intrahepatic bile duct dilatation in the anterior segment of the right hepatic lobe. Bronchoscopy revealed bile in the bronchial tree. Percutaneous transhepatic cholangiogram demonstrated a fistula from the bile duct of the anterior segment of the right lobe of the liver to the right lower lobe of the lung (Fig 2). Initially, she was treated with external biliary stents which decreased symptoms. However, when the biliary stents were occluded, bilious sputum would increase.
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Fig 1. Computed tomography demonstrating consolidation of the lateral basal segment of the right lobe.
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Fig 2. Percutaneous transhepatic cholangiogram demonstrating the fistula from the anterior segment of the right lobe of the liver to the right lower lobe of the lung.
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Two months later, the patient underwent a right thoracotomy to repair the bronchobiliary fistula. At thoracotomy, the anterior segment of the right lower lobe which communicated with the biliary system was resected. The diaphragmatic defect measured 1.5 cm in diameter. The defect was obliterated with a vascularized intercostal muscle pedicle; the diaphragm was closed primarily around the muscle. The patient had an uneventful recovery and the biliary stents were intermittently clamped postoperatively. However, the patient developed fever, abdominal pain, and elevated liver function tests with clamping. She has required continued stenting due to recurrent pain from biliary ductal strictures and stones. At last follow-up 10 months from her thoracotomy, she has had no further bilioptysis.
Patient 2
A 61-year-old male underwent a right hepatectomy for carcinosarcoma of the right lobe of the liver. His postoperative course was uncomplicated, and he was discharged on the 6th postoperative day. Six days later, he presented at his home hospital with a right empyema and subdiaphragmatic abscess. Cultures demonstrated Enterobacter cloacae. A percutaneous abdominal drain was placed with CT guidance with resolution of the subdiaphragmatic fluid collection. Following 2 weeks of intravenous antibiotic treatment, he was discharged from his home hospital. One week later, the catheter was removed, only to have the patient develop rigors and a productive cough 3 weeks later. Further evaluation at his home hospital revealed reaccumulation of the subdiaphragmatic abscess, intrahepatic fluid collection, and a density in the right lower lung. He was again managed with percutaneous abdominal drainage and antibiotics. Over the next few months, the abscess cavity had decreased in size but the infiltrate in the lung persisted. Bilioptysis developed, and he was referred to our institution.
Endoscopic retrograde cholangiopancreatography demonstrated a fistula between the biliary tree and the right lower lobe of the lung (Fig 3). At thoracotomy, a portion of the involved right lower lobe was excised which demonstrated chronic inflammation. The catheter in the biliary system was injected, but a fistula was not found. The area of inflammation on the diaphragm was debrided and covered with a pericardial fat pad. The patient did well postoperatively and has had no recurrence of bilioptysis after 7 months of follow-up.
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Fig 3. Endoscopic retrograde cholangiopancreatography demonstrating communication between the biliary system with the bronchial tree. Percutaneously placed drain exiting the right flank.
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Comment
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Several mechanisms can result in bronchobiliary fistulae. One mechanism involves biliary obstruction which produces an inflammatory reaction in the subdiaphragmatic space with subsequent rupture into the bronchial system. Another mechanism is when hydatid cysts erode through the diaphragm into the pleural cavity and adjacent bronchial tree, thus creating a fistula.
Diagnosis is based on clinical presentation. Patients often present with bilioptysis which is intermittent bile-stained sputum. In addition, acute bronchiolitis from the intense inflammatory reaction of the bronchial mucosa secondary to bile or even chronic bronchopneumonia can be present [1]. Time of inciting event to patient presentation is also variable, ranging from weeks to months. When the diagnosis is in doubt, the presence of bilirubin in a sputum sample is diagnostic. Most fistulae involve the right basilar segments of the lung.
Defining the biliary anatomy and the corresponding fistulous tract is helpful to plan management. Roentgenographic imaging includes percutaneous transhepatic cholangiography or endoscopic retrograde cholangiopancreatography. CT and ultrasound may also be used to delineate associated pathology (ie, subphrenic abscess, empyema). Mazzuca and associates have suggested cholescintigraphy with 99mTc-diethyl-IDA for fistula visualization [2].
Treatments for bronchobiliary fistulae have traditionally been surgical, with simple drainage of the subdiaphragmatic abscess with or without resection of the fistulous tract and involved lung [3, 4]. With the advent of minimally invasive surgery, endoscopic placement of stents to relieve the obstruction have been reported [5]. Others have also used endoscopic sphincterotomy in addition to a stent for treatment of the bronchobiliary fistula [6–8]. These reports have introduced a nonsurgical treatment to a disease process that has traditionally been treated surgically.
However, these less invasive treatment modalities usually involve prolonged hospitalization and multiple roentgenographic manipulations. Despite multiple attempts at noninvasive therapies, the patient ultimately returns to the operating room for more definitive treatment. Both of our patients were managed with a surgical resection of the fistula. The abdominal pathology was treated with biliary diversion via stents to relieve the obstruction. As an adjunct to resection, viable tissue was interposed between the lung and the diaphragmatic defect. Tissue used for the interposition most frequently is muscle or fat.
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References
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Gugenheim J., Ciardullo M., Traynor O., Bismuth H. Bronchobiliary fistulas in adults. Ann Surg 1988;207:90-94.[Medline]
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Mazzuca N., Giulianotti P.C., Mosca F., Toni M.G., Bianchi R., Mariani G. Visualization of a bronchobiliary fistula during cholescintigraphy with 99mTc-diethyl-IDA. J Nucl Med Allied Sci 1983;27:257-260.[Medline]
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Johnson M.M., Chin R., Jr, Haponik E.F. Thoracobiliary fistula. South Med J 1996;89:335-339.[Medline]
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Cropper L.D., Gold R.E., Roberts L.K. Bronchobiliary fistula. J Trauma 1982;22:68-70.[Medline]
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D’Altorio R.A., McAllister J.D., Sestric G.B., Cichon P.J. Hepatopulmonary fistula. Am J Gastroenterol 1992;87:784-786.[Medline]
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Yilmaz U., Sahin B., Hilmioglu F., Tezel A., Boyacioglu S., Cumhur T. Endoscopic treatment of bronchobiliary fistula. Hepatogastroenterology 1996;43:293-300.[Medline]
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Khandelwal M., Inverso N., Conter R., Campbell D. Endoscopic management of bronchobiliary fistula. J Clin Gastroenterol 1996;23:125-127.[Medline]
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Moreira V.F., Arocena C., Cruz F., Alvarez M., San Roman A.L. Bronchobiliary fistula secondary to biliary lithiasis. Dig Dis Sci 1994;39:1994-1999.[Medline]
Accepted for publication January 23, 2000.
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Abstract
Introduction
