Komatsu Shohei, Iwasaki Takeshi, Nishioka Naritomo, Toyokawa Akihiro, Teramura Kazuhiro
Department of Surgery, Yodogawa Christian Hospital, Osaka, Japan.
Department of Surgery, Yodogawa Christian Hospital, Osaka, Japan; Department of Surgery, National Hospital Organization, Kobe Medical Center, Hyogo, Japan.
Ann Vasc Surg. 2014 Nov;28(8):1934.e13-7. doi: 10.1016/j.avsg.2014.06.078. Epub 2014 Jul 11.
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.
胆道出血是胃肠道出血的一种罕见且可能致命的病因。虽然肝动脉动脉瘤是胆道出血的一个病因，但非创伤性病例鲜有报道。在此，我们描述一例患有马凡综合征的患者因反复胆道出血而需要肝切除术的巨大肝动脉动脉瘤病例。一名53岁男性因突发上腹部疼痛和黄疸前来我院就诊。腹部计算机断层扫描显示肝门部有一个巨大的肝动脉动脉瘤，急诊内镜逆行胆管造影显示胆道出血。鉴于动脉瘤导致了胆道出血，我们进行了腹部血管造影以进行治疗。该检查显示一个血栓形成的动脉瘤以及左肝动脉周围迂曲的异常血管，这些异常血管似乎环绕着动脉瘤。因此，我们栓塞了左肝动脉，立即实现了止血。此后又发生了3次再出血，每次均进行了经动脉栓塞，虽能迅速止血但只是暂时的。然后，进行了急诊左半肝切除术并切除了动脉瘤。切除标本的病理检查显示动脉瘤完全血栓形成且机化；然而，在动脉瘤壁和胆管之间有异常小动脉增生。该病例的独特之处在于，导致胆道出血的是机化的肝动脉动脉瘤诱发的异常小动脉，而非动脉瘤本身。
