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成人心血管型庞贝病患者升主动脉巨大动脉瘤:组织学表现类似纤维连接素病。

Huge aneurysm of the ascending aorta in a patient with adult-type Pompe's disease: histological findings mimicking fibrillinopathy.

机构信息

Department of Cardiovascular Surgery, Inselspital, University Hospital, University of Bern, Bern, Switzerland.

出版信息

Eur J Cardiothorac Surg. 2013 Jan;43(1):193-5. doi: 10.1093/ejcts/ezs489. Epub 2012 Sep 3.

Abstract

Adult-type Pompe's disease (glycogen storage disease type II) has rarely been shown to present with dilatative arteriopathy, suggesting potential smooth muscle involvement in addition to lysosomal glycogen deposits usually restricted to skeletal muscle tissue. We report the case of a middle-aged man under enzyme replacement therapy presenting with an exceedingly large thoracic aortic aneurysm. Surprisingly, the histological work-up of resected aortic tissue revealed changes mimicking those observed in patients with classic connective tissue diseases. Enzyme replacement therapy, in addition to musculoskeletal and pulmonary treatment for patients with Pompe's disease, may prolong survival and lead to patients presenting with vascular alterations that may pose surgical and potential diagnostic challenges in the future.

摘要

成人型庞贝病(糖原贮积病 II 型)很少表现为扩张性动脉病,这表明除了通常局限于骨骼肌组织的溶酶体糖原沉积外,平滑肌也可能受累。我们报告了一例接受酶替代治疗的中年男性,他患有非常大的胸主动脉瘤。令人惊讶的是,切除的主动脉组织的组织学检查显示出类似于经典结缔组织疾病患者的改变。除了庞贝病患者的肌肉骨骼和肺部治疗外,酶替代治疗还可能延长生存时间,并导致患者出现血管改变,这可能在未来带来手术和潜在诊断方面的挑战。

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