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通过外显子组测序鉴定一个患有I型并指畸形c型的中国家系中的错义HOXD13突变。

Identification of a missense HOXD13 mutation in a Chinese family with syndactyly type I-c using exome sequencing.

作者信息

Deng Hao, Tan Ting, He Quanyong, Lin Qiongfen, Yang Zhijian, Zhu Anding, Guan Liping, Xiao Jingjing, Song Zhi, Guo Yi

机构信息

Center for Experimental Medicine, The Third Xiangya Hospital, Central South University, Changsha, Hunan 410013, P.R. China.

Department of Burn and Plastic Surgery, The Third Xiangya Hospital, Central South University, Changsha, Hunan 410013, P.R. China.

出版信息

Mol Med Rep. 2017 Jul;16(1):473-477. doi: 10.3892/mmr.2017.6576. Epub 2017 May 11.

Abstract

Syndactyly is one of the most common hereditary limb malformations, and is characterized by the fusion of specific fingers and/or toes. Syndactyly type I‑c is associated with bilateral cutaneous or bony webbing of the third and fourth fingers and occasionally of the third to fifth fingers, with normal feet. The aim of the present study was to identify the genetic basis of syndactyly type I‑c in four generations of a Chinese Han family by exome sequencing. Exome sequencing was conducted in the proband of the family, followed by direct sequencing of other family members of the same ancestry, as well as 100 ethnically‑matched, unrelated normal controls. A missense mutation, c.917G>A (p.R306Q), was identified in the homeobox D13 gene (HOXD13). Sanger sequencing verified the presence of this mutation in all of the affected family members. By contrast, this mutation was absent in the unaffected family members and the 100 ethnically‑matched normal controls. The results suggest that the c.917G>A (p.R306Q) mutation in the HOXD13 gene, may be responsible for syndactyly type I‑c in this family. Exome sequencing may therefore be a powerful tool for identifying mutations associated with syndactyly, which is a disorder with high genetic and clinical heterogeneity. The results provide novel insights into the etiology and diagnosis of syndactyly, and may influence genetic counseling and the clinical management of the disease.

摘要

并指畸形是最常见的遗传性肢体畸形之一,其特征是特定手指和/或脚趾融合。I-c型并指畸形与第三和第四指的双侧皮肤或骨性蹼有关,偶尔第三至五指也有,足部正常。本研究的目的是通过外显子组测序确定一个中国汉族家庭四代人中I-c型并指畸形的遗传基础。对该家庭的先证者进行外显子组测序,随后对同一家族的其他家族成员以及100名种族匹配的无关正常对照进行直接测序。在家obox D13基因(HOXD13)中鉴定出一个错义突变,c.917G>A(p.R306Q)。桑格测序证实所有受影响的家庭成员中都存在这种突变。相比之下,未受影响的家庭成员和100名种族匹配的正常对照中不存在这种突变。结果表明,HOXDl3基因中的c.917G>A(p.R306Q)突变可能是该家族I-c型并指畸形的原因。因此,外显子组测序可能是识别与并指畸形相关突变的有力工具,而并指畸形是一种具有高度遗传和临床异质性的疾病。这些结果为并指畸形的病因学和诊断提供了新的见解,并可能影响该疾病的遗传咨询和临床管理。

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