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[法布里病的分子病理学与临床表现]

[Molecular pathology and clinical manifestations of Fabry disease].

作者信息

Rákóczi Eva, Görögh Sándor, Grubits János, Erdos Melinda, Garzuly Ferenc, Hahn Katalin, Bencsik Krisztina, Vécsei László, Trinn Csilla, Kristóf Eva, Mogyorósy Gábor, Tóth Beáta, Maródi László

机构信息

Debreceni Egyetem, Orvos- és Egészségtudományi Centrum Infektológiai és Gyermekimmunológiai Tanszék Debrecen Nagyerdei krt. 98. 4012, Hungary.

出版信息

Orv Hetil. 2007 Jun 10;148(23):1087-94. doi: 10.1556/OH.2007.28086.

Abstract

Fabry disease is a rare, progressive lysosomal storage disorder caused by mutation in the GAL gene and an impaired function of the alpha-galactosidase A enzyme. The enzymatic defect results in the progressive accumulation of glycosphingolipids in endothelial cells, smooth muscle cells, leucocytes and fibroblasts leading to organ damage in the skin, eye, nervous system, kidney and heart. Major clinical manifestations include acroparesthesis, angiokeratoma, corneal opacities, vascular diseases of the heart, kidney, and the central nervous system. Enzyme replacement therapy has recently become available for the treatment of Fabry patients. In this review the authors describe clinical features of Fabry disease in 31 Hungarian patients. At the time of this analysis the database consisted of 31 cases (15 males, 16 females) of whom 5 have died (4 males, 1 female). The most common disease-specific manifestation was angiokeratoma in males, and eye symptoms in females. 25% of female subjects were symptom free. Genotyping was performed in all cases and disease-causing mutations were found in all families. Three new mutations were identified. Twelve patients (8 males and 4 females) are currently receiving enzyme replacement therapy.

摘要

法布里病是一种罕见的进行性溶酶体贮积症,由GAL基因突变和α-半乳糖苷酶A酶功能受损引起。酶缺陷导致糖鞘脂在内皮细胞、平滑肌细胞、白细胞和成纤维细胞中进行性蓄积,从而导致皮肤、眼睛、神经系统、肾脏和心脏的器官损害。主要临床表现包括肢端感觉异常、血管角质瘤、角膜混浊、心脏、肾脏和中枢神经系统的血管疾病。酶替代疗法最近已可用于治疗法布里病患者。在这篇综述中,作者描述了31例匈牙利患者的法布里病临床特征。在进行此次分析时,数据库包含31例病例(15例男性,16例女性),其中5例已死亡(4例男性,1例女性)。最常见的疾病特异性表现是男性的血管角质瘤和女性的眼部症状。25%的女性受试者无症状。对所有病例进行了基因分型,所有家族均发现了致病突变。鉴定出三个新突变。12例患者(8例男性和4例女性)目前正在接受酶替代疗法。

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