Bronchobiliary fistula caused after hepatectomy for hepatocellular carcinoma: a case report
© The Author(s). 2016
Received: 9 August 2016
Accepted: 1 December 2016
Published: 5 December 2016
A bronchobiliary fistula, an intercommunication between the biliary tract and bronchial trees, is an extremely rare complication after hepatectomy.
A 70-year-old male underwent partial resection of the liver for recurrent hepatocellular carcinoma under a thoracoabdominal approach. The immediate postoperative clinical course was uneventful, but the patient was febrile and laboratory examinations revealed leukocytosis on the 15th postoperative day. An intraabdominal abscess was suspected based on the computed tomography findings, and percutaneous drainage was performed. Bile was drained, and fluoroscopy using a contrast medium from the drainage tube revealed a communication between the cavity and the common hepatic duct. Two weeks after drainage, bilioptysis was seen. Fistulography demonstrated the presence of the bronchus in the right lower lobe of the lung via the subphrenic space. Therefore, the patient was diagnosed to have a bronchobiliary fistula. Fistulography revealed closure of the communication with the bronchus about a month after drainage. However, the bile leakage and bilioptysis did not stop even after endoscopic nasogastric biliary drainage, and ethanol injection therapy were performed. Eventually, residual right bisectionectomy without resection of the fistulous tract and involved lung was performed to remedy the intractable bile leakage. The clinical course after the reoperation was good without bile leakage, bilioptysis, or pulmonary disorders, and the patient was discharged 40 days after reoperation.
We experienced a rare case of bronchobiliary fistula that occurred after hepatectomy for hepatocellular carcinoma. Careful attention should be paid to prevent bile leakage during hepatectomy, since bile leakage has the potential to cause a bronchobiliary fistula.
KeywordsBronchobiliary fistula Bile leakage Hepatectomy
A bronchobiliary fistula (BBF), an intercommunication between the biliary tract and bronchial trees, is a rare condition. Several causes of BBF, including amoebic diseases of the liver, trauma, biliary obstruction, and hepatobiliary surgery, have been reported, and appropriate treatment is required, because a BBF can be associated with high mortality and morbidity rates . We herein present a case required reoperation for BBF with intractable bile leakage that occurred after partial hepatic resection for hepatocellular carcinoma.
However, bile continued to be discharged at a rate of 50 to 100 ml per day, and it became clear that this was caused by an injured bile duct in segment 5 of the liver by fistulography and drip infusion cholangiography-CT 3 months after the initiation of drainage. At that time, the hilar side of the bile duct could not be demonstrated by fistulography (Fig. 3c), and the periphery of the injured bile duct could also not be demonstrated by endoscopic retrograde cholangiography (ERC) (Fig. 3d). ERC revealed no stricture of the common hepatic duct or common bile duct. Based on these findings, the bile leakage in this patient was thought to have lapsed into the interrupted type due to continuous drainage.
Review of reported cases with bronchobiliary fistula after treatments for hepatocellular carcinoma
Location of HCC
Treatment for HCC
Biloma or abscess
Disorder of biliary Tree
Diagnostic modality of BBF
Treatment for BBF (prognosis)
(5 months; died of cancer)
(5 months; died of cancer)
PTAD and RML+RLL
(12 days; died of hepatic failure)
Biliary stent and RHx+RLL+BR
(6 months; alive)
Lateral segment and dome
TACE and RFA
(2 months; alive)
(2 months; alive)
VATS (pneumolysis and resection of the BBF) (1 year; alive)
Thoracic cavity drainage
(1 day; died of respiratory failure)
(18 months; alive)
Bronchoscopy and tubogram
Drainage and embolization of the BBF (1 month; died of hepatic failure)
(40 days; alive)
Segment 5, 7
(4 years; alive)
A BBF is thought to be a serious complication associated with a high morbidity rate, including high rates of sepsis and pulmonary disorders, and often results in death . Therefore, appropriate and prompt treatment is required for BBF, but there has been no consensus-based standard treatment. A few reports demonstrated that a BBF could be healed using only percutaneous drainage with the administration of antibiotics [7, 19]. However, most reported cases of BBF required additional treatment. Less-invasive procedures, such as endoscopic biliary drainage and placement of biliary stents have recently been employed in the treatment of BBF, especially in cases associated with biliary tract obstruction, because the endoscopic techniques have been improved. Surgical approaches with or without resection of the fistula tract and involved lung should be considered only after other intervention have failed [17, 20, 21]. BBF of our present case seemed to be not exactly healed only by continuous percutaneous drainage because bilioptysis was often seen, although bronchus was not demonstrated by fistulography. It was speculated that our patient could not recover from BBF as long as bile leakage was persistent.
Bile leakage is one of the most common complications after hepatic resection, and around 70% of the cases of bile leakages are thought to resolve spontaneously . However, the bile leakage in the present case could not be healed by long-term percutaneous drainage. Several treatments, including ENBD and ethanol injection therapy, were attempted, but failed. Eventually, the nonsurgical procedures were concluded to be of limited use, and residual anterior sectionectomy of the liver, including the leaking ducts, was planned preoperatively based on volumetric analysis (ratio of estimated hepatic resection volume 22.1%) because BBF believed to be healed if bile leakage stopped. However, residual right bisectionectomy was actually performed because residual anterior sectionectomy might cause bile leakage although the patient had a risk of the postoperative hepatic failure due to major hepatectomy. Fortunately, the postoperative clinical course was good without bile leakage, bilioptysis, or pulmonary disorders.
In conclusion, we experienced a rare case of BBF that occurred after hepatectomy for hepatocellular carcinoma. Careful attention should be paid to prevent bile leakage during hepatectomy since bile leakage has the potential to cause a BBF.
SH and YI drafted the manuscript. YI performed the surgery and SH, TH, KS, and AY participated in the surgery. JF comprehensively supervised this study. All authors read and approved the final manuscript.
The authors declare that they have no competing interests.
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Ethics approval and consent to participate
The authors declare that they have no ethical conflicts.
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- Jamal Y, Tombazzi C, Waters B, Ismail MK. Bronchobiliary fistula in a cirrhotic patient: a case report and review of the literature. Am J Med Sci. 2008;335:315–9.View ArticlePubMedGoogle Scholar
- Peacock TB. Case in which hydatids were expectorated and one of suppuration of a hydatid cyst of the liver communicating with the lungs. Edin Med J. 1850;74:33–46.Google Scholar
- Khandelwal M, Inverso N, Conter R, Campbell D. Endoscopic management of a bronchobiliary fistula. J Clin Gastroenterol. 1996;23:125–7.View ArticlePubMedGoogle Scholar
- Akazawa S, Omagari K, Amenomori M, Nishiyama H, Mizuta Y, Kohno S. Bronchobiliary fistula associated with intrahepatic biloma after transcatheter arterial chemoembolization for hepatocellular carcinoma. J Hepatol. 2004;40:1045–6.View ArticlePubMedGoogle Scholar
- Kaido T, Kano M, Suzaki S, Yanagibashi K, Shiota M. Bronchobiliary fistula after hepatectomy for hepatocellular carcinoma. Dig Dis Sci. 2006;51:1117–21.View ArticlePubMedGoogle Scholar
- Hibi T, Sakamoto Y, Asamura H, Tochigi N, Ojima H, Shimada K, et al. Successful resection of hepatocellular carcinoma with bronchobiliary fistula caused by repeated transcatheter arterial embolizations: report of a case. Surg Today. 2007;37:154–8.View ArticlePubMedGoogle Scholar
- Yoon DH, Shim JH, Lee WJ, Kim PN, Shin JH, Kim KM. Percutaneous management of a bronchobiliary fistula after radiofrequency ablation in a patient with hepatocellular carcinoma. Korean J Radiol. 2009;10:411–5.View ArticlePubMedPubMed CentralGoogle Scholar
- Kim DH, Choi DW, Choi SH, Heo JS, Jeong J, Rhu J. Surgical treatment of bronchobiliary fistula due to radiofrequency ablation for recurrent hepatocellular carcinoma. Korean J Hepatobiliary Pancreat Surg. 2013;17:135–8.View ArticlePubMedPubMed CentralGoogle Scholar
- Kuo YS, Lee SC, Chang H, Hsieh CB, Huang TW. Thoracoscopic surgery for bronchobiliary fistula: a case report. J Cardiothorac Surg. 2014;9:139.View ArticlePubMedPubMed CentralGoogle Scholar
- Dai H, Cui D, Li D, Zhai BO, Zhang J. Hepatic abscess with hepatobronchial fistula following percutaneous radiofrequency ablation for hepatocellular carcinoma: a case report. Oncol Lett. 2015;9:2289–92.PubMedPubMed CentralGoogle Scholar
- Zhong Y, Deng M, Li K, Xu R. Delayed bronchobiliary fistula and cholangiolithiasis following percutaneous radio frequency ablation for hepatocellular carcinoma. Exp Biol Med (Maywood). 2015;240:156–9.View ArticleGoogle Scholar
- Kim HY, Kwon SH, Oh JH, Shin JS, Dong SH, Park MJ, et al. Percutaneous transhepatic embolization of a bronchobiliary fistula developing secondary to a biloma after conventional transarterial chemoembolization in a patient with hepatocellular carcinoma. Cardiovasc Intervent Radiol. 2016;39:628–31.View ArticlePubMedGoogle Scholar
- Zeng Z, Cai M, Huang W, Huang J, Chen X, Shan H, et al. Delayed bronchobiliary fistula following radiofrequency ablation in a patient with hepatocellular carcinoma: a case report and lesson regarding treatment. Oncol Lett. 2016;11:3213–7.PubMedPubMed CentralGoogle Scholar
- Gugenheim J, Ciardullo M, Traynor O, Bismuth H. Bronchobiliary fistulas in adults. Ann Surg. 1988;207:90–4.View ArticlePubMedPubMed CentralGoogle Scholar
- Chua HK, Allen MS, Deschamps C, Miller DL, Pairolero PC. Bronchobiliary fistula: principles of management. Ann Thorac Surg. 2000;70:1392–4.View ArticlePubMedGoogle Scholar
- Koch KA, Crump JM, Monteiro CB. A case of biliptysis. J Clin Gastroenterol. 1995;20:49–53.View ArticlePubMedGoogle Scholar
- Liao GQ, Wang H, Zhu GY, Zhu KB, Lv FX, Tai S. Management of acquired bronchobiliary fistula: a systematic literature review of 68 cases published in 30 years. World J Gastroenterol. 2011;17:3842–9.View ArticlePubMedPubMed CentralGoogle Scholar
- Oettl C, Schima W, Metz-Schimmerl S, Fugger R, Mayrhofer T, Herold CJ. Bronchobiliary fistula after hemihepatectomy: cholangiopancreaticography, computed tomography and magnetic resonance cholangiography findings. Eur J Radiol. 1999;32:211–5.View ArticlePubMedGoogle Scholar
- Cropper Jr LD, Gold RE, Roberts LK. Bronchobiliary fistula: management with percutaneous catheter drainage of a subphrenic abscess. J Trauma. 1982;22:68–70.View ArticlePubMedGoogle Scholar
- Rose DM, Rose AT, Chapman WC, Wright JK, Lopez RR, Pinson CW. Management of bronchobiliary fistula as a late complication of hepatic resection. Am Surg. 1998;64:873–6.PubMedGoogle Scholar
- Aydin U, Yazici P, Tekin F, Ozutemiz O, Coker A. Minimally invasive treatment of patients with bronchobiliary fistula: a case series. J Med Case Rep. 2009;3:23.View ArticlePubMedPubMed CentralGoogle Scholar
- Vigano L, Ferrero A, Sgotto E, Tesoriere RL, Calgaro M, Capussotti L. Bile leak after hepatectomy: predictive factors of spontaneous healing. Am J Surg. 2008;196:195–200.View ArticlePubMedGoogle Scholar