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X连锁低汗性外胚层发育不良:新特征及一种新的EDA基因突变

X-Linked Hypohidrotic Ectodermal Dysplasia: New Features and a Novel EDA Gene Mutation.

作者信息

Savasta Salvatore, Carlone Giorgia, Castagnoli Riccardo, Chiappe Francesca, Bassanese Francesco, Piras Roberta, Salpietro Vincenzo, Brazzelli Valeria, Verrotti Alberto, Marseglia Gian L

机构信息

Department of Pediatrics, Fondazione Policlinico San Matteo IRCCS, University of Pavia, Pavia, Italy.

出版信息

Cytogenet Genome Res. 2017;152(3):111-116. doi: 10.1159/000478922. Epub 2017 Sep 7.

Abstract

We described a 5-year-old male with hypodontia, hypohidrosis, and facial dysmorphisms characterized by a depressed nasal bridge, maxillary hypoplasia, and protuberant lips. Chromosomal analysis revealed a normal 46,XY male karyotype. Due to the presence of clinical features of hypohidrotic ectodermal dysplasia (HED), the EDA gene, located at Xq12q13.1, of the patient and his family was sequenced. Analysis of the proband's sequence revealed a missense mutation (T to A transversion) in hemizygosity state at nucleotide position 158 in exon 1 of the EDA gene, which changes codon 53 from leucine to histidine, while heterozygosity at this position was detected in the slightly affected mother; moreover, this mutation was not found in the publically available Human Gene Mutation Database. To date, our findings indicate that a novel mutation in EDA is associated with X-linked HED, adding it to the repertoire of EDA mutations.

摘要

我们描述了一名5岁男性,患有牙齿发育不全、少汗症以及面部畸形,其特征为鼻梁凹陷、上颌骨发育不全和嘴唇突出。染色体分析显示核型为正常的46,XY男性。由于存在少汗性外胚层发育不良(HED)的临床特征,对该患者及其家族位于Xq12q13.1的EDA基因进行了测序。先证者序列分析显示,EDA基因外显子1中核苷酸位置158处存在半合子错义突变(T到A颠换),该突变使密码子53由亮氨酸变为组氨酸,而在症状较轻母亲的该位置检测到杂合性;此外,在公开的人类基因突变数据库中未发现此突变。迄今为止,我们的研究结果表明,EDA基因中的一种新突变与X连锁HED相关,这为EDA基因突变库增添了新内容。

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