Spontaneous Rupture of an Intrahepatic Biliary Duct Causing a Right Pleural and Retroperitoneal Biloma

Abstract

Bilomas are encapsulated collections of bile outside of the biliary tree and are mainly caused by iatrogenic injury or abdominal trauma. Spontaneous rupture of the biliary tree is not common. We report a case of a patient with spontaneous rupture biloma treated with percutaneous and internal-external biliary drainage. A 68-year-old man with significant history of amyloidosis and monoclonal paraproteinemia was admitted for progressively worsening right upper quadrant abdominal pain and nausea for the last week. Vital signs were unremarkable and physical exam revealed mild jaundice and a positive Murphy sign. No rebound or guarding. Laboratory results revealed total bilirubin of 1.7 mg/dL, direct bilirubin of 0.8 mg/dL, alkaline phosphatase of 328 IU/L, AST of 190 IU/L, ALT 154 IU/L. Pancreatic enzymes and Ca 19-9 were within normal limits. Right upper quadrant ultrasound showed a distended gallbladder with biliary sludge and a dilated common bile duct (CBD) measuring 11mm. MRCP suggested a pancreatic head mass. Subsequent ERCP showed narrowing at the CBD making it difficult for cannulation. However fine needle aspiration samples were obtained and came back positive for malignancy. Over the next few days, the patient became febrile, continued having the abdominal pain and developed worsening liver function tests. CT of the abdomen revealed a large retroperitoneal fluid collection, intra and extrahepatic biliary duct dilation and a right pleural effusion. Patient underwent right chest tube placement, which drained 2 L of bilious pleural fluid. She also underwent percutaneous transhepatic cholangiogram, which showed bilious leakage from third order branch of the right hepatic duct at the level of the superior aspect of the right lobe of the liver, which was causing complete obstruction of the distal CBD. Subsequent internal-external biliary drain was placed without complications, followed by resolution of the patient's jaundice over the next days. Follow up CT of the abdomen confirmed resolution of the retroperitoneal and right pleural bilomas. After discharge, patient underwent Whipple procedure for resection of what was found to be a well differentiated cholangiocarcinoma of the CBD. He has been following with general surgery and hematology/oncology since then. Spontaneous biloma is a rare occurrence which needs to be promptly assessed and treated. Percutaneous biliary drainage with placement of an internal-external drain is an effective procedure which can be used, especially if endoscopic sphincterotomy is not feasible.