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一名临床疑似洛伊茨-迪茨综合征患者因巨大假性动脉瘤导致胰胆管狭窄的内镜治疗

Endoscopic management of pancreatic and biliary duct stenoses due to a giant pseudoaneurysm in a patient clinically suggestive of Loeys-Dietz syndrome.

作者信息

Tanikawa Tomohiro, Akagi Akihisa, Kawada Mayuko, Ishii Katsunori, Fushimi Takashi, Urata Noriyo, Suehiro Mitsuhiko, Shiraha Hidenori, Haruma Ken, Kawamoto Hirofumi

机构信息

Department of General Internal Medicine 2, Kawasaki Medical School, 2-6-1, Nakasange, Kita-Ku, Okayama, 700-8505, Japan.

Department of General Surgery, Kawasaki Medical School, 2-6-1, Nakasange, Kita-Ku, Okayama, 700-8505, Japan.

出版信息

Clin J Gastroenterol. 2025 May 28. doi: 10.1007/s12328-025-02151-w.

Abstract

Loeys-Dietz syndrome is a rare connective tissue disorder characterized by the formation of aggressive arterial aneurysms. There are a few reports of Loeys-Dietz syndrome with pseudoaneurysms causing simultaneous pancreatic and biliary stenoses. Herein, we report the case of a 42-year-old man with Loeys-Dietz syndrome who presented with acute pancreatitis and liver dysfunction caused by a giant pancreaticoduodenal artery pseudoaneurysm compressing the main pancreatic and bile ducts. To minimize the risk of pseudoaneurysm rupture during endoscopic intervention, transcatheter arterial embolization was performed, followed by endoscopic intervention. Although initial clinical improvement was observed after endoscopic stent placement, a fistula between the pancreatic duct and the thrombosed pseudoaneurysm was detected at 4 months but spontaneously closed with continued stenting. Despite persistent ductal stenosis requiring long-term stent management, the fistula had closed 1 year after the initial stent placement. To the best of our knowledge, this is the first report describing a structured treatment strategy for pancreaticobiliary obstruction caused by a Loeys-Dietz syndrome-related pseudoaneurysm. This case highlights the importance of a stepwise interventional radiology-first approach and careful follow-up for the management of complex vascular compressive syndromes.

摘要

洛伊迪茨综合征是一种罕见的结缔组织疾病,其特征是形成侵袭性动脉动脉瘤。有少数关于洛伊迪茨综合征伴假性动脉瘤导致同时性胰腺和胆管狭窄的报道。在此,我们报告一例42岁患有洛伊迪茨综合征的男性患者,其因巨大的胰十二指肠动脉假性动脉瘤压迫主胰管和胆管而出现急性胰腺炎和肝功能障碍。为了将内镜干预期间假性动脉瘤破裂的风险降至最低,先进行了经导管动脉栓塞,然后进行内镜干预。尽管在内镜支架置入后观察到最初的临床改善,但在4个月时检测到胰管与血栓形成的假性动脉瘤之间存在瘘管,但通过持续置入支架瘘管自发闭合。尽管持续性导管狭窄需要长期支架管理,但在最初置入支架1年后瘘管已闭合。据我们所知,这是第一份描述针对洛伊迪茨综合征相关假性动脉瘤引起的胰胆管梗阻的结构化治疗策略的报告。该病例强调了逐步以介入放射学为先的方法以及对复杂血管压迫综合征进行仔细随访管理的重要性。

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