CASE REPORT Spontaneous hepatic subcapsular biloma: Report of three cases with review of the literature Majid MUSHTAQUE1, Mohammad FAROOQ MIR2, Parvez NAZIR2, Parwez Sajad KHAN1, Vidhi DADU3, Lateef A DAR1, Rayees A DAR1, Samina A KHANDAY2 Departments of 1General Surgery and 2Radiodiagnosis & Imaging, Sher-i Kashmir Institute of Medical Sciences, Saura, Srinagar, Jamnu and Kashmir, India Department of 3Radiodiagnosis & Imaging, Urmila Dadu Diagnostic Centre, Modhi Nagar, U.P., INDIA, U.P., India A biloma is an encapsulated bile collection outside the biliary tree. Most cases are caused mainly by iatrogenic injury and trauma, and are usually located in the sub-hepatic space. Spontaneous biloma is an uncommon entity. We report three rare cases of sponta- neous hepatic subcapsular biloma formation in association with choledocholithiasis in two patients and cholangiocarcinoma in one patient. All the patients presented with extrahepatic biliary obstruction with no previous history of abdominal surgery, instrumen- tation, or trauma. Ultrasound and computerized tomography of the abdomen documented hepatic subcapsular biloma. The patients were initially managed with antibiotics and radiologically guided pigtail drainage of the collections, followed by definitive treat- ment of their underlying cause. Key words: Biloma, hepatic, subcapsular, choledocholithiasis, cholangiocarcinoma Spontan hepatik subkapsüler bilioma: Üç vakan›n sunumu ve literatürün gözden geçirilmesi Bilioma, safra yolu d›fl›nda yerleflen kapsüllü safra kolleksiyonudur. Bunlar›n büyük ço¤unlu¤u iatrojenik nedenlere veya travma- ya ba¤l› olarak geliflir ve genellikle subhepatik bofllukta yerleflirler. Spontan bilioma nadir görülen bir durumdur. Burada nadir görülen, ikisi koledokolitiyazise ve biri kolanjiokarsinoma sekonder geliflen spontan hepatik subkapsüler bilioma vakas› sunulmufl- tur. Tüm hastalar ekstrahepatik biliyer obstrüksiyon ile baflvurmufltur ve hiçbirinde abdominal cerrahi öyküsü, enstrümentasyon veya travma öyküsü yoktur. Hepatik subkapsüler biliomalar›n tan›mlanmas›nda ultrasonografi ve bilgisayarl› tomografi kullan›l- m›flt›r. Hastalar ilk olarak antibiyotik ve radjoloji klavuzlu¤unda pig-tail katater drenaj› ile tedavi edilmifl ve ertesinde alt yatan etkenin özgül tedavisi yap›lm›flt›r. Anahtar kelimeler: Bilioma, hepatik, subkapsüler, koledokolitiyazis, kolanjiyokarsinoma INTRODUCTION The term “biloma”, denoting an encapsulated ex- Non-traumatic bilomas are commonly referred to trahepatic collection of bile, was first introduced as spontaneous bilomas. The most common un- by Gould and Patel in 1979 (1). The main causati- derlying cause of spontaneous biloma is choledoc- ve factors of biloma formation are traumatic or iat- holithiasis (3). Other rare causes include bile duct rogenic injuries, including abdominal surgery, tumors, hepatic infarction and idiopathic. The percutaneous catheter drainage, transhepatic cho- mechanism of spontaneous biloma formation is langiogram, and endoscopic retrograde cholangi- thought to be either the raised intraductal pressu- opancreatography (ERCP), and infrequently wit- re secondary to extrahepatic biliary obstruction hout an apparent cause (2). (EHBO), rupture of a cyst/diverticulum, or a focal Address for correspondence: Majid MUSHTAQUE Manuscript received: 23.12.2010 Accepted: 22.03.2011 Department of General Surgery, Sher-i Kashmir Institute of Medical Sciences, Turk J Gastroenterol 2012; 23 (3): 284-289 doi: 10.4318/tjg.2012.0346 Saura, Srinagar, Jamnu and Kashmir, India Phone: +919 419 010 280 E-mail: [email protected] Spontaneous hepatic subcapsular biloma liver infarction (3, 4). We report three cases of ting, fever, and yellowish discoloration of the eyes spontaneously formed hepatic subcapsular bilo- of seven days’ duration. There was no significant mas secondary to EHBO, and we discuss how to medical history and no history of abdominal sur- diagnose and manage such patients, together with gery, instrumentation or trauma. She was jaundi- a review of the related literature. ced and febrile. There were no respiratory or car- diovascular abnormalities. Abdominal examinati- CASE REPORTS on revealed scratch marks on the abdominal wall, with mild diffuse tenderness. There were no other Case 1 abnormalities on the examination. Initial investi- A 55-year-old man was admitted with intermittent gations revealed a white cell count of 13,500/mm3 colicky abdominal pain, fever and jaundice. He had no previous history of abdominal surgery, instru- mentation or trauma. His general physical exami- a nation revealed icterus. On the abdominal exami- nation, there was mild tenderness in the right up- per abdomen with no palpable lump or organome- galy. Prophylactic broad spectrum antibiotics were started on admission. Laboratory investigations revealed: hemoglobin: 12.6 g/dl, white blood cells: 12,500/mm3 (87% neutrophils), blood urea: 33 mg/dl, and creatinine: 1.2 mg/dl. Liver function test revealed a total bilirubin of 10 mg/dl with a conjugated component of 5.6 mg/dl; aspartate ami- notransferase (AST) and alanine aminotransferase (ALT) were mildly raised. Alkaline phosphatase (ALP) was grossly increased (1060 mg/dl). The chest X-ray revealed right plural effusion and a raised right hemidiaphragm. Ultrasonography b (USG) of the abdomen revealed a large unilocular subcapsular fluid collection measuring 9 x 8 x 5 cm, going around the right lobe of the liver antero- laterally and reaching the bare area. There was no free fluid in the abdominal cavity. The common bi- le duct (CBD) was 13 mm in diameter with a hype- rechoic shadow at its lower part. Intrahepatic bili- ary radicals (IHBR) were also dilated. Thoracoab- dominal computed tomography (CT) confirmed a large hepatic subcapsular biloma with volume of approximately 230 ml (Figure 1a, 1b) with a CT number of 18 Hounsfield units (HU). Based on the imaging findings, the patient was subjected to USG-guided pigtail catheter drainage of the collec- tion, which drained about 160 ml of bile during the first 24 hours (h). Minimal drainage was observed over the next few days. Endoscopic sphincterotomy was done, and a 13 mm solitary stone was retrie- ved. The subcapsular fluid collection resolved, and the external percutaneous pigtail catheter was re- moved after five days. No residual or recollection Figure 1a and 1b. Contrast-enhanced computed tomography was seen on follow-up USG at two and six weeks. (CECT) of the abdomen showing a large unilocular subcapsular Case 2 fluid collection around the right lobe of the liver anterolaterally and reaching up to the bare area with a volume of 230 ml. Com- A 65-year-old woman was admitted with compla- mon bile duct (CBD) and intrahepatic biliary radicals (IHBR) ints of abdominal pain, nausea, intermittent vomi- are dilated. 285 MUSHTAQUE et al. with neutrophilia (91%). Other blood counts were the 10th day after a normal T-tube cholangiogram. otherwise normal as were blood urea, serum cre- On follow-up, USG of the abdomen did not show atinine and electrolytes. Liver function tests re- any residual or recollection of bile. vealed: bilirubin: 9.5 mg/dl (normal: 0.3-1.0 mg/dl) Case 3 with direct component of 5.5 mg/dl, ALP: 680 (nor- mal: 20-90 IU/L), AST: 180 U/L (normal: 5-45 A 50-year-old female presented with right upper U/L), ALT: 290 U/L (normal: 5-40 U/L), and gam- abdominal pain and progressive jaundice of three ma-glutamyl transpeptidase (GGT): 87 U/L (nor- weeks’ duration. There was also a history of nause- mal: <65 U/L), with a normal albumin and coagu- a, fatigue, anorexia, and weight loss. The patient logram. Ultrasound examination done at a perip- had no history of abdominal surgery, instrumenta- heral hospital had reported ascites. USG of the ab- tion or trauma. On general physical examination, domen was repeated in our hospital, which revea- she had mild pallor and icterus. Abdominal exami- led a huge hepatic subcapsular collection exten- nation revealed mild tenderness in the right ding all around the liver and causing severe com- hypochondrium with a palpable gallbladder. Car- pression of the liver parenchyma and IHBR. A lar- diovascular and respiratory examinations were in- ge 18 mm hyperechoic shadow was seen in the dis- conclusive. Complete blood count revealed only tal CBD. Contrast-enhanced CT (CECT) of the ab- anemia (hemoglobin 8 g/dl). Liver function test re- domen confirmed a massive hepatic subcapsular vealed a total bilirubin of 12 mg/dl with a conjuga- collection of approximately 1500 ml (Figure 2a, 2b) ted component of 7 mg/dl; AST and ALT were mild- with a CT number of 24 HU. CT-guided pigtail ly raised, while ALP and GGT were grossly increa- catheter drainage of the collection was done, sed. Coagulogram was mildly deranged with inter- which removed about 1200 ml of altered bile in the national normalized ratio (INR) of 1.4. Other labo- first 2 h after the procedure. The patient was re- ratory parameters were normal. USG of the abdo- lieved of acute pain immediately. The bile culture men revealed a subcapsular hepatic collection with revealed a mixed growth of coli forms. The patient dilated CBD and IHBR. On CECT of the abdomen, was already on broad spectrum antibiotics. Mini- diffuse enhancing thickening of the lower CBD was mal drainage was noted after the first 48 h. ERCP noticed with abrupt cut-off at its lower end, along revealed a dilated CBD and two stones (largest 20 with a large hepatic subcapsular collection predo- mm) visible in the lumen. CBD exploration with minantly posteromedially along segment V1 of the choledocholithotomy and T-tube drainage was do- liver. It was about 11 x 10 x 5 cm in size with a vo- ne, as ERCP and sphincterotomy failed to manage lume of approximately 300 ml and a CT number of the choledocholithiasis. The pigtail catheter was 16 HU (Figure 3a, 3b). Broad spectrum antibiotics removed during surgery. The postoperative period were started, and CT-guided pigtail catheter dra- was uneventful, and the T-tube was removed on inage of the collection was done, which removed a b Figure 2a and 2b. CECT of the abdomen revealed a huge hepatic subcapsular collection (1500 ml) extending all around the liver and causing severe compression of the liver parenchyma and IHBR.