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一名西班牙患者的病例报告,该患者患有致心律失常性右心室心肌病和掌跖角化病,无桥粒斑蛋白和桥粒芯蛋白基因修饰。

Case report of a Spanish patient with arrhythmogenic right ventricular cardiomyopathy and palmoplantar keratoderma without plakoglobin and desmoplakin gene modifications.

作者信息

Alonso-Orgaz Sergio, Zamorano-León Jose J, Fernandez-Arquero Miguel, Villacastín Julian, Perez-Castellanos Nicasio, García-Torrent Maria J, Macaya Carlos, López Farré Antonio J

出版信息

Int J Cardiol. 2007 May 31;118(2):275-7. doi: 10.1016/j.ijcard.2006.06.065. Epub 2006 Oct 12.

DOI:10.1016/j.ijcard.2006.06.065
PMID:17045679
Abstract

We report a case of a 43 year old man from Spain, who has been diagnosed with Naxos disease. It is a hereditary disorder characterized by palmoplantar keratoderma, woolly hair and cardiomyopathy, which has been associated with a mutation in plakoglobin encoding gene in chromosome 17q21. In the patient, the direct sequencing of the plakoglobin gene discarded TG deletion at 2157 characteristic of Naxos disease. Analysis of the reported desmoplakin mutations associated with Carvajal Syndrome, another ARVC disease, that it is also accompanied with a skin and hair disorder, also failed to reveal mutations in desmoplakin gene. These results suggest the existence of other causative genes and/or other putative sites in desmoplakin/plakoglobin encoding genes than those recently published.

摘要

我们报告了一例来自西班牙的43岁男性病例,该患者被诊断患有纳克索斯病。这是一种遗传性疾病,其特征为掌跖角化病、羊毛状发和心肌病,与17q21染色体上的桥粒芯蛋白编码基因突变有关。在该患者中,桥粒芯蛋白基因的直接测序排除了纳克索斯病特有的2157位TG缺失。对与另一种致心律失常性右室心肌病(ARVC)疾病——卡瓦哈尔综合征相关的报道的桥粒斑蛋白突变进行分析,该疾病也伴有皮肤和毛发疾病,结果也未发现桥粒斑蛋白基因存在突变。这些结果表明,除了最近公布的那些基因外,在桥粒斑蛋白/桥粒芯蛋白编码基因中还存在其他致病基因和/或其他假定位点。

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