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血管型埃勒斯-当洛斯综合征表现为搏动性颈部肿块:病例报告及文献复习。

Vascular Ehlers-Danlos Syndrome Presenting as a Pulsatile Neck Mass: a Case Report and Review of Literature.

机构信息

Department of Medicine, University of California, San Francisco Medical Center, San Francisco, CA, USA.

出版信息

J Gen Intern Med. 2018 Jul;33(7):1192-1195. doi: 10.1007/s11606-018-4445-3. Epub 2018 Apr 26.

Abstract

Ehlers-Danlos syndrome refers to a spectrum of connective tissue disorders typically caused by mutations in genes responsible for the synthesis of collagen. Patients with Ehlers-Danlos syndrome often exhibit hyperflexibility of joints, increased skin elasticity, and tissue fragility. Vascular Ehlers-Danlos (vEDS) is a subtype of Ehlers-Danlos syndrome with a predilection to involve blood vessels. As such, it often manifests as vascular aneurysms and vessel rupture leading to hemorrhage. There are few reports describing primary prevention of aneurysms in the setting of undiagnosed, suspected vEDS. We present a case of a 30-year-old woman who presents with a pulsatile neck mass found to have multiple arterial aneurysms on imaging, hyperflexibility, and characteristic facial features consistent with vEDS. As described in this case, management of a suspected connective tissue disorder is a multidisciplinary approach including vascular surgery, medical therapy, and genetic testing to confirm the diagnosis. We review literature regarding the care of patients with vascular Ehlers-Danlos as it might pertain to hospitalized patients.

摘要

埃勒斯-当洛斯综合征是一种结缔组织疾病谱,通常由负责胶原合成的基因突变引起。埃勒斯-当洛斯综合征患者常表现为关节过度活动、皮肤弹性增加和组织脆弱。血管型埃勒斯-当洛斯(vEDS)是埃勒斯-当洛斯综合征的一种亚型,易累及血管。因此,它常表现为血管动脉瘤和血管破裂导致出血。目前很少有报告描述在未确诊、疑似 vEDS 的情况下对动脉瘤进行一级预防。我们报告了一例 30 岁女性,因发现颈部搏动性肿块就诊,影像学检查发现多处动脉动脉瘤,同时存在关节过度活动和特征性面部表现,符合 vEDS 的特征。正如本病例所述,疑似结缔组织疾病的管理是一种多学科方法,包括血管外科、药物治疗和基因检测以确认诊断。我们回顾了关于血管型埃勒斯-当洛斯患者护理的文献,因为这可能与住院患者有关。

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