Rare cause of obstructive haemobilia with recurrent biliopancreatic complications: a paradigmatic case

Abstract

Haemobilia is an unusual but significant cause of upper gastrointestinal bleeding. Two-thirds of haemobilia cases are secondary to invasive hepato-biliopancreatic procedures. Biliary angiodysplasia is exceptionally unusual, with only three cases reported. Herein, we report the case of an autonomous 80-year-old woman with a history of cholecystectomy 5 years ago and cardiovascular disease—hypertension, heart failure, acute myocardial infarction, stroke and non-valvular atrial fibrillation, anticoagulated with apixaban 2.5 mg two times per day. Since July 2019, she had four episodes of acute cholangitis of mild-to-moderate severity, having undergone broad spectrum antibiotics treatment and endoscopic retrograde cholangiopancreatography (ERCP), with sphincterotomy and bile sludge extraction. After 3 months, the patient presented with a new episode of acute cholangitis, this time with haemobilia (Quincke’s triad). An abdominal CT angiography showed no evidence of active bleeding, with plastic biliary prosthesis left by ERCP. The patient continued presenting new episodes of acute cholangitis with haemobilia, some of them with associated pancreatitis. A cholangioscopy with Spyglass DS II was performed, showing an angiodysplasia occupying half of the luminal circumference of the middle choledoccus, without active haemorrhage. After a multidisciplinary meeting and given the high haemorrhagic/thrombotic risk (CHA2DS2-VASc 8), closure of the left atrial appendage was considered. However, relapse of the condition after beginning the antiaggregation protocol for cardiovascular intervention made it unfeasible. Another cholangioscopy with an ultra-thin endoscope for argon-plasma coagulation was attempted, without success. The abdominal CT angiography was repeated, this time with identification of dilated ramifications of the gastroduodenal and inferior pancreatic arteries. After embolisation of these aberrant vessels with microcoils, the patient went well, with no recurrence of bleeding or biliopancreatic complications. We present a case of obstructive haemobilia with multiple biliopancreatic complications, secondary to an extremely rare cause—choledochal angiodysplasia. Cholangioscopy had a decisive role in the diagnosis and therapeutic guidance. The diagnostic/therapeutic challenge associated with haemobilia stands out, with the need for a personalised and multidisciplinary approach.