Gastroenterology, Centro Hospitalar e Universitario de Coimbra EPE, Coimbra, Portugal
Gastroenterology, Centro Hospitalar e Universitario de Coimbra EPE, Coimbra, Portugal.
BMJ Case Rep. 2022 Feb 25;15(2):e245303. doi: 10.1136/bcr-2021-245303.
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.
肝内淤血是上消化道出血的一个不常见但很重要的原因。三分之二的肝内淤血病例继发于侵入性肝胆胰手术。胆管血管发育不良非常罕见,仅报告过三例。在此,我们报告一例自主 80 岁女性,5 年前行胆囊切除术,患有心血管疾病-高血压、心力衰竭、急性心肌梗死、中风和非瓣膜性心房颤动,每日两次口服阿哌沙班 2.5 毫克抗凝。自 2019 年 7 月以来,她经历了四次轻度至中度急性胆管炎发作,接受了广谱抗生素治疗和内镜逆行胰胆管造影术(ERCP),括约肌切开术和胆汁淤积物抽吸。3 个月后,患者出现新的急性胆管炎发作,伴有肝内淤血(Quincke 三联征)。腹部 CT 血管造影显示无活动性出血,ERCP 后留下塑料胆道支架。患者继续出现新的伴有肝内淤血的急性胆管炎发作,其中一些伴有胰腺炎。进行了Spyglass DS II 胆管镜检查,显示中胆管腔周长的一半被血管发育不良占据,无活动性出血。经过多学科会议,考虑到出血/血栓形成高风险(CHA2DS2-VASc 8),考虑行左心耳封堵术。然而,心血管介入抗聚集方案开始后病情复发,导致该方案不可行。尝试使用超细内镜进行氩等离子凝固的另一次胆管镜检查,但未成功。再次进行腹部 CT 血管造影,发现胃十二指肠和胰下动脉扩张分支。用微线圈栓塞这些异常血管后,患者情况良好,无再发出血或胆胰并发症。我们报告一例因极其罕见的胆总管血管发育不良引起的阻塞性肝内淤血伴多种胆胰并发症的病例。胆管镜检查在诊断和治疗指导方面具有决定性作用。肝内淤血的诊断/治疗挑战突出,需要个性化和多学科方法。