Respiratory Medicine CME (2008) 1, 164 168 respiratory MEDICINE CME CASE REPORT Bronchobiliary fistula treated with histoacryl embolization under bronchoscopic guidance: A case report Jung Hyun Kim a, Man Deuk Kim b, Young Kyung Lee b, Seong Gyu Hwang c, Ji Hyun Lee a, Eun Kyung Kim a, Hye Cheol Jeong a, a Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Bundang CHA Hospital, Pochon CHA University, 351 Yatap-dong, Sungnam City, Kyonggi-do 463-712, South Korea b Department of Radiology, Bundang CHA Hospital, Pochon CHA University, 351 Yatap-dong, Sungnam City, Kyonggi-do 463-712, South Korea c Division of Gastroenterology and Hepatology, Bundang CHA Hospital, College of Medicine, Pochon CHA University, 351 Yatap-dong, Sungnam City, Kyonggi-do 463-712, South Korea Received 12 March 2008; accepted 11 April 2008 KEYWORDS Bronchobiliary fistula; Bronchoscopy; Therapeutic embolization Summary A 56-year-old woman presented with bile-tinged sputum for one year. Five years ago, she was diagnosed with hepatocellular carcinoma. She was treated with five hepatic arterial chemotherapies, two radiofrequency ablations, four chemoembolizations, and a right lobectomy of the liver. Two years ago, a liver abscess with biliary stricture occurred and was treated with endoscopic biliary drainage and balloon dilatation. On admission, bronchoscopy suggested a bronchobiliary fistula in the anterobasal segment of the right lower lobar bronchus. Fluorography showed a fistulous tract between the right lower lobar bronchus and the liver abscess pocket. We performed histoacryl embolization under bronchoscopic guidance and the bile-tinged sputum resolved. However, biliary obstruction and hepatic failure followed and the patient expired three months later. We report a case of a bronchobiliary fistula managed with a bronchoscopic approach. & 2008 Elsevier Ltd. All rights reserved. Introduction Corresponding author. Tel.: +82 31 780 6142; fax: +82 31 4800. E-mail address: jhcmd@hanmail.net (H.C. Jeong). A bronchobiliary fistula (BBF) is a rare complication following liver disease, surgery, procedures, or trauma. Most therapeutic approaches aim to lower biliary tract pressure. 1 In cases of bronchopleural fistulae, bronchoscopic 1755-0017/$ - see front matter & 2008 Elsevier Ltd. All rights reserved. doi:10.1016/j.rmedc.2008.04.002
Bronchoscopic embolization of bronchobiliary fistula 165 interventions have been tried, sometimes. However, only one case of a bronchoscopic approach has been reported for BBF. 2 We report a case of BBF after resection of a hepatoma treated with histoacryl embolization under bronchoscopic guidance. Case report A 56-year-old woman presented with bile-tinged sputum for one year. Twenty years before presentation, she was diagnosed as a hepatitis B virus carrier. Five years before presentation, she was diagnosed with liver cirrhosis and hepatocellular carcinoma in segments VI and VII (Figure 1). Because there was portal vein thrombosis, she received intra-hepatic arterial chemotherapy and 2340 cgy of external radiation therapy. After five cycles of hepatic arterial chemotherapy with cisplatin and 5-fluorouracil, computed tomography revealed decreased hepatocellular carcinoma and resolved portal vein thrombosis. The patient underwent two treatments of radiofrequency ablation (RFA) (Figure 1) and transcatheter arterial chemoembolization (TACE). Two years before presentation, she underwent surgical right lobectomy for recurrent hepatocellular carcinoma. Two months after surgery, biliary obstruction occurred and repeated endoscopic retrograde biliary drainage (ERBD) with plastic stent was performed (Figure 2), but the biliary obstruction was not resolved. Six months after surgery, a liver abscess with stent failure was diagnosed and treated with percutaneous abscess drainage and percutaneous transhepatic biliary drainage (PTBD) (Figure 3). Eleven months before presentation, bile-tinged sputum occurred and an immediate surgical fistulectomy was performed. After that, repeated PTBD and percutaneous balloon dilatation (PBD) for biliary stricture were performed (Figure 4), but bilioptysis persisted and three months before presentation she was admitted for recurrent pneumonia. On admission, she was not febrile. She complained of bilecolored sputum and cough in the morning. Her vital signs were normal. Physical examination showed crackles on the right lower lung field. Chest radiography revealed haziness on the right lower lung field and pleural effusion in the right hemithorax. There was no dysfunction in the percutaneous biliary drainage system. A complete blood count revealed mild anemia (hemoglobin of 9.4 g/dl). Blood chemistry showed a total bilirubin of 3.95 mg/dl and an elevated alkaline phosphatase of 260 IU/ L. The prothrombin time was 17.5 s. Bronchoscopy showed bile-colored material in the anterobasal segment of the right lower lobar bronchus (Figure 5). Figure 1 Liver CT of five years ago: (a) arterial phase reveals a huge mass at segments VI and VII. Portal vein thrombosis is present (arrow) and (b) after arterial chemotherapy and radiofrequency ablation. Figure 2 Biliary obstruction at two years before presentation. Endoscopic retrograde biliary drainage with plastic stent was performed.
166 J.H. Kim et al. Figure 3 Follow-up CTscan six months after surgery: (a) CT scan revealed liver abscess and (b) after percutaneous drainage, abscess pocket decreased. Figure 4 Cholangiogram via endoscopic approach. (a) Cholangiogram showed abscess pocket (arrow) and bronchus (arrowhead) is visualized through fistulous tract. (b) Percutaneous biliary balloon dilatation was performed. Figure 5 bronchus. Bronchoscopy revealed bile-colored material in the sub-segment of the anterobasal segment of the right lower lobar A chest computed tomography with multiplanar reconstruction suggested a BBF in the anterobasal and lateral basal segments (Figure 6). Fluorography under bronchoscopic guidance revealed a fistulous tract with an anterobasal segment of the right lower lobar bronchus and an abscess pocket in the liver (Figure 7). A guidewire was inserted via the bronchoscope channel into the anterobasal segment of the right lower lobe. A 5-Fr Berenstein catheter was passed over the guidewire. A microcatheter was placed into the abscess pocket of the liver. The abscess pocket and fistula tract were embolized with a mixture of 1 cc of histoacryl and 2 cc of lipiodol (Figure 7).
Bronchoscopic embolization of bronchobiliary fistula 167 After embolization, bilioptysis stopped and the chest radiograph showed a decreased extent of haziness in the right lower lung field (Figure 8). Although bilioptysis was resolved, biliary obstruction and bile peritonitis occurred. Three months later, she expired due to hepatic failure. Discussion Figure 6 The chest CT with multiplanar reconstruction. Consolidation in the anterobasal segment of the right lower lobe with peripheral bronchiectasis that communicated directly with the pleural space. A BBF is a rare complication of liver disease, trauma, procedures, or surgery. The most common cause of a BBF is a liver abscess. 3 In the United States, the most common cause of BBF is biliary obstruction. 4 Definitive treatment for a BBF is a surgical fistulectomy with soft tissue reconstruction. However, the surgical approach is accompanied by significant morbidity, mortality, and frequent reoperations. 3 Recently, conservative interventions have been the preferred procedure. Most interventions aim to lower biliary tract pressure; ERBD or PTBD is the preferred approaches. 1 Most patients respond well to these approaches, but it takes time for healing and some patients do not respond. There is one reported case of BBF treated with n-butyl cyanoacrylate (NBCA) embolization via a bronchial approach. 2 In that case, a BBF was developed after right hepatic Figure 7 Histoacryl embolization: (a) fluoroscopic image revealed abscess pocket (arrowhead) and fistulous tract (arrow), (b) dye was drained into biliary tree, and (c) after histoacryl emboliztion. Figure 8 Chest radiograph before (a) and after (b) embolization procedure.
168 lobectomy for single colon cancer metastasis. A 60% NBCA and 40% ethiodol mixture (3 cc) was used for embolization. In our case, BBF developed after lobectomy for hepatocellular carcinoma. Despite successful percutaneous biliary dilatation and PTBD, the patient complained of persistent bile-tinged sputum and recurrent pneumonia. We used histoacryl and lipiodol for embolization material. Tissue glue via a bronchoscopic approach is often used for bronchopleural fistulae. 5 In those cases, bronchoscopic intervention had an acceptable success rate without serious complications. The patient with colon cancer was free from symptoms for three months and died of cancer progression. 2 Our patient was also free from bilioptysis, but biliary obstruction and hepatic failure followed. Embolization under bronchoscopic guidance can be an effective option for BBF; however, one should pay attention to possible complications. References J.H. Kim et al. 1. Khandelwal M, Inverso N, Conter R, et al. Endoscopic management of a bronchobiliary fistula. J Clin Gastroenterol 1996;23: 125 7. 2. Goldman SY, Greben CR, Setton A, et al. Bronchobiliary fistula successfully treated with n-butyl cyanoacrylate via a bronchial approach. J Vasc Interv Radiol 2007;18:151 5. 3. Borrie J, Shaw JHF. Hepatobronchial fistula caused by hydatid disease. The Dunedin experience 1952 79. Thorax 1981;36:25 8. 4. Rose DM, Rose AT, Chapman WC, et al. Management of bronchobiliary fistula as a late complication of hepatic resection. Am Surg 1998;64:873 6. 5. West D, Togo A, Kirk AJ. Are bronchoscopic approaches to postpneumonectomy bronchopleural fistula an effective alternative to repeat thoracostomy. Interact Cardiovasc Thorac Surg 2007;6: 547 50. Conflict of interest statement The authors have no competing interests to declare. The manuscript has been reviewed and approved by all authors.