INTRODUCTION

True visceral aneurysms (VA) are rare with an incidence of 0.1% to 2%.1Hepatic artery aneurysms (HAA) are one of the most common VA, with 60% of them located in the common hepatic artery, 30% in the right hepatic artery, and 5% in the left hepatic artery.2–6 Because of its rarity, HAA are often diagnosed late, and symptoms can range from being asymptomatic to life-threatening hemorrhage from rupture or formation of a fistula. Rarely, HAA can cause arteriobiliary fistula, also known as common hepatic artery-bile duct fistula (CBDF), leading to gastrointestinal hemorrhage. Immediate and effective treatment is crucial in such cases, given the rapid evolution toward hemodynamic instability. We report a case of CBDF successfully treated with endoscopic and surgical intervention.

CASE REPORT

A 61-year-old man with a medical history of alcohol and tobacco use disorder presented with 4 days of melena and epigastric pain. On arrival, the patient was noted to be hypotensive, and initial laboratory data revealed a white blood cell count 8.7 g/dL, hemoglobin 8.0 g/dL (baseline 12.5 g/dL), alkaline phosphatase 183 U/L, aspartate aminotransferase 204 U/L, alanine aminotransferase 204 U/L, and total bilirubin 4.4 mg/dL. The initial computed tomography (CT) of the abdomen was suggestive of HAA which prompted CT angiography. The CT angiography revealed a prominent 47 × 33-mm fusiform aneurysm of the common hepatic artery and a fistulous tract into the porta hepatis. The aneurysm was compressing the common bile duct, and there was noted to be blood in the bile duct, raising suspicion for CBDF (Figure 1).

Figure 1.:

(A, B) CTA showing a prominent 47 × 33-mm fusiform aneurysm of the CHA and a fistulous tract into the porta hepatis, compressing the CBD with hemobilia (red arrow). CBD, common bile duct; CHA, common hepatic artery; CTA, computed tomography angiography.

Vascular surgery was consulted, and an urgent esophagogastroduodenoscopy was performed to rule out any other possible source of bleeding. Esophagogastroduodenoscopy revealed active bleeding from the major papilla further raising concern for CBDF (Figure 2). After a multidisciplinary discussion with hepatobiliary surgery and vascular surgery, the patient underwent emergent vascular surgical repair with supraceliac aortohepatic bypass. Exploration of the aneurysm sac revealed arterial bleeding with mural thrombus and bile leak. Because of concern of bile leak, the patients underwent endoscopic retrograde cholangiopancreatography (ERCP) with intraoperative cholangiogram, which revealed a leak at the proximal and distal end of the HAA repair. A 10 × 180-mm fully covered self-expanding metal stent was deployed, which resulted in the clearance of profuse bleeding (Figure 3). His liver function tests subsequently normalized, and the leak resolved on subsequent ERCP 3 months later. The patient did well clinically and was discharged the following day. The fully covered self-expanding metal stent was successfully removed 3 months later (Figure 3).

Figure 2.:

EGD showing bleeding from the major papilla. EGD, esophagogastroduodenoscopy.

Figure 3.:

(A, B) Fluoroscopic image revealing biliary leak (red arrow) and subsequent fully covered metal stent placement.

DISCUSSION

HAA are a rare phenomenon, representing most VA.7,8 True HAA are mostly due to degenerative or dysplastic change of the extrahepatic vessels. The most common causes include atherosclerosis, fibromuscular dysplasia, cystic medial necrosis, and portal hypertension. Less common etiologies include autoimmune disease, collagen vascular diseases, hypertension, congenital disorders, trauma, and inflammatory conditions.4,9 Clinical presentation can range from being asymptomatic to gastrointestinal hemorrhage, right upper-quadrant abdominal pain, and obstructive jaundice, also known as Quincke's triad. In asymptomatic individuals, repair should be considered when the aneurysm is greater than 2 cm. Larger aneurysms have a 25% risk of rupture and have a 70% mortality on rupture.7,8 An important complication of HAA is CBDF, which can oftentimes lead to life-threatening gastrointestinal hemorrhage.

CBDF causing hemobilia has been reported sparingly in the literature. Most cases are a result of iatrogenic trauma, with the advent of invasive procedures such as percutaneous liver biopsy, transhepatic cholangiography, biliary drainage, and other conditions such as arteritis or infection.10 A case by Páez-Carpio et al11 reported an arteriobiliary fistula caused by an aneurysm in a patient with liver transplantation who had undergone an ERCP for stenosis of the bile duct anastomosis site. CBDF has also been reported after percutaneous transhepatic biliary drainage and open urgent cholecystectomy.12,13 In both cases, the patients were treated with surgery and stenting. Interestingly, our patient had no previous instrumentation or trauma to explain the CBDF, suggesting the idiopathic nature of this rare etiology.

Diagnosis of CBDF is based on cross-sectional imaging. Optimal imaging requires contrasted CT or magnetic resonance imaging of the mesenteric vasculature.14 Given the paucity of cases, there are no definitive treatment guidelines for CBDF. Management requires multidisciplinary discussion with radiology, gastroenterology, vascular surgery, and hepatobiliary surgery. Achieving hemostasis, maintenance of bile flow, and stabilization of acute hemorrhage remains crucial in treatment. Surgical management along with endovascular repair and stenting has shown promising results, as was seen in our case.

CBDF is a rare presentation that can lead to life-threatening hemorrhage. Patients presenting with hemobilia, abdominal pain, and jaundice should raise suspicion for CBDF. Given the associated morbidity and mortality, early recognition can lead to prompt diagnosis and initiation of life-saving treatment.

DISCLOSURES

Author contributions: H. Chaudhry and Y. Ichkhanian reviewed the literature, drafted the manuscript, revised it for important intellectual content, and were involved in the final approval of the version to be published. SO Salvador and J. Yang revised the article for important intellectual content and were involved in the final approval of the version to be published. H. Chaudhry is the article guarantor.

Financial disclosure: None to report.

Previous presentation: This case was presented at the American College of Gastroenterology Virtual Annual Scientific Meeting; October 2020.

Informed consent was obtained for this case report.

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