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康拉迪-许纳曼-哈普勒综合征中的两种新型EBP突变。

Two novel EBP mutations in Conradi-Hünermann-Happle syndrome.

作者信息

Ausavarat Surasawadee, Tanpaiboon Pranoot, Tongkobpetch Siraprapa, Suphapeetiporn Kanya, Shotelersuk Vorasuk

机构信息

Division of Medical Genetics and Metabolism, Department of Pediatrics, Faculty of Medicine, King Chulalongkorn Memorial Hospital, Bangkok, Thailand.

出版信息

Eur J Dermatol. 2008 Jul-Aug;18(4):391-3. doi: 10.1684/ejd.2008.0433. Epub 2008 Jun 23.

Abstract

Conradi-Hünermann-Happle syndrome, also known as chondrodysplasia punctata type 2 (CDPX2), is an X-linked dominant disorder characterized by skin defects, skeletal and ocular abnormalities. CDPX2 was shown to be caused by mutations in the gene encoding emopamil binding protein (EBP). At least 58 different mutations have been described. Here we present clinical and molecular findings in two unrelated Thai girls with CDPX2. Mutation analysis by PCR-sequencing the entire coding region of EBP successfully revealed two potentially pathogenic, novel mutations, c.616G-->T and c.382delC. This study has expanded the spectrum of the EBP gene mutations causing CDPX2.

摘要

康拉迪-许纳曼-哈普尔综合征,也称为2型点状软骨发育不良(CDPX2),是一种X连锁显性疾病,其特征为皮肤缺陷、骨骼和眼部异常。已证明CDPX2是由编码埃莫帕米结合蛋白(EBP)的基因突变引起的。已描述了至少58种不同的突变。本文报告了两名患CDPX2的不相关泰国女孩的临床和分子学研究结果。通过对EBP整个编码区进行PCR测序进行突变分析,成功发现了两个潜在致病的新突变,即c.616G→T和c.382delC。本研究扩大了导致CDPX2的EBP基因突变谱。

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