Hemobilia associated with a giant thrombosed aneurysm of the hepatic artery requiring hepatectomy.

Hemobilia is an unusual and potentially catastrophic cause of gastrointestinal bleeding. Although hepatic artery aneurysm is a cause of hemobilia, nontraumatic cases are infrequently reported. Herein, we describe the case of a giant hepatic artery aneurysm requiring hepatectomy because of repeated hemobilia in a patient with Marfan syndrome. A 53-year-old man presented to our hospital with sudden epigastric pain and jaundice. Abdominal computed tomography showed a giant hepatic arterial aneurysm in the porta hepatis, and emergency endoscopic retrograde cholangiography revealed hemobilia. Assuming that the aneurysm caused the hemobilia, we performed an abdominal angiogram for treatment. The study revealed a thrombosed aneurysm along with tortuous abnormal vessels in the periphery of the left hepatic artery, which appeared to surround the aneurysm. Therefore, we embolized the left hepatic artery, and immediate hemostasis was achieved. Rebleeding occurred 3 times thereafter, and each time, transarterial embolization was performed, resulting in prompt but only temporary hemostasis. Then, emergency left hemihepatectomy and resection of the aneurysm were performed. Pathologic examination of the resected specimen revealed that the aneurysm was completely thrombosed and organized; however, abnormal arterioles proliferated between the aneurysmal wall and the bile duct. The unique feature of this case was that the abnormal arterioles induced by the organized hepatic artery aneurysm, not the aneurysm itself, caused the hemobilia.