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台湾地区克氏综合征患者的突变分析。

Mutation analysis of Crouzon syndrome in Taiwanese patients.

作者信息

Chang Chin-Ping, Wan Lei, Tsai Chang-Hai, Lee Cheng-Chun, Tsai Fuu-Jen

机构信息

Department of Medical Research, China Medical University Hospital, Taichung, Taiwan.

出版信息

J Clin Lab Anal. 2006;20(1):23-6. doi: 10.1002/jcla.20096.

Abstract

Crouzon syndrome is an autosomal-dominant disorder that causes premature fusion of the cranial suture. Crouzon, Pfeiffer, and Apert syndromes are caused by mutations in the extracellular, third immunoglobulin-like domain, and adjacent linker regions (exons IIIa and IIIc) of the fibroblast growth factor receptor 2 (FGFR2) gene. We screened 12 Crouzon syndrome patients for mutations in exons IIIa and IIIc of the FGFR2 gene by polymerase chain reaction (PCR) and direct sequencing. Mutations were detected in nine of 12 patients at amino acid positions 278, 281, 289, 342, and 354. More than half of the studied Crouzon patients carried a mutation resulting in either the loss or gain of a cysteine residue. A novel missense Ser354Phe substitution at exon IIIc of the human FGFR2 gene was found. According to our results, sequencing analysis of IgIII domain of the FGFR2 gene can lead to a genetic diagnosis of Crouzon syndrome.

摘要

克鲁宗综合征是一种常染色体显性疾病,可导致颅缝过早融合。克鲁宗综合征、普费弗综合征和阿佩尔综合征是由成纤维细胞生长因子受体2(FGFR2)基因的细胞外第三免疫球蛋白样结构域及相邻连接区(外显子IIIa和IIIc)发生突变引起的。我们通过聚合酶链反应(PCR)和直接测序,对12例克鲁宗综合征患者的FGFR2基因外显子IIIa和IIIc的突变情况进行了筛查。在12例患者中的9例中检测到了位于氨基酸位置278、281、289、342和354处的突变。超过半数的被研究克鲁宗患者携带导致半胱氨酸残基缺失或增加的突变。发现了人类FGFR2基因外显子IIIc处一个新的错义突变Ser354Phe。根据我们的研究结果,对FGFR2基因IgIII结构域进行测序分析可实现对克鲁宗综合征的基因诊断。

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