When Cholangitis Reveals Liver Involvement in Hereditary Hemorrhagic Telangiectasia: A Case Report

胆管炎揭示遗传性出血性毛细血管扩张症的肝脏受累:病例报告

阅读:1

Abstract

Hereditary hemorrhagic telangiectasia (HHT) syndrome is a rare autosomal dominant hereditary disorder characterized by multiple arteriovenous malformations (AVMs), resulting from capillary dysplasia, responsible for arteriovenous shunting. It can affect various organs, including the lungs, liver, and central nervous system. Clinical manifestations are epistaxis, telangiectasias, and complications of visceral AVMs. Biliary involvement is uncommon and typically results from biliary ischemia. This can lead to complications such as cholangitis, biliary strictures, cysts, necrosis, and bilomas. Although less frequent, biliary complications may present similarly to bile duct obstruction or sepsis and necessitate careful management. We report the case of a 55-year-old patient with a history of unexplored epistaxis who presented with hepatic colic. Clinical and laboratory findings revealed telangiectasias, cholestasis, and an inflammatory syndrome. Imaging revealed hepatic artery dilatation, multiple AVMs in hepatic telangiectasias and a porto-hepatic shunt, and segmental cystic dilation of the bile ducts. The patient was treated for cholangitis and remains under surveillance. This case highlights the importance of multimodal imaging in evaluating hepatic involvement in patients with HHT. Indeed, a combination of ultrasound, multiphasic CT, and dynamic MRI helps reveal not only typical vascular abnormalities, including hepatic artery dilatation, telangiectasias, and arteriovenous shunts, but also biliary involvement. Regular follow-up is necessary to tailor management and prevent potential complications associated with this hereditary vascular syndrome.

特别声明

1、本页面内容包含部分的内容是基于公开信息的合理引用;引用内容仅为补充信息,不代表本站立场。

2、若认为本页面引用内容涉及侵权,请及时与本站联系,我们将第一时间处理。

3、其他媒体/个人如需使用本页面原创内容,需注明“来源:[生知库]”并获得授权;使用引用内容的,需自行联系原作者获得许可。

4、投稿及合作请联系:info@biocloudy.com。