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新发现的 GLI1 基因杂合序列变异导致轴后多指(趾)畸形。

Novel heterozygous sequence variant in the GLI1 underlies postaxial polydactyly.

机构信息

Department of Chemistry, University of Azad Jammu and Kashmir, Muzaffarabad, Pakistan.

Department of Biochemistry, Faculty of Biological Sciences, Quaid-i-Azam University, Islamabad, Pakistan.

出版信息

Congenit Anom (Kyoto). 2020 Jul;60(4):115-119. doi: 10.1111/cga.12361. Epub 2019 Oct 29.

Abstract

Polydactyly is one of the most common congenital abnormal phenotype of autopod, which is characterized by extra supernumerary digit in hands/feet with or without well-developed bony structure within the digits. Preaxial polydactyly (PPD), postaxial polydactyly (PAP), and meso-axial (central) polydactyly are three different isoforms of polydactyly. Genetically, at least 10 genes have been identified causing nonsyndromic polydactyly. In the present study, we have investigated a large family segregating autosomal dominant form of nonsyndromic polydactyly. Whole exome sequencing followed by Sanger sequencing revealed a novel heterozygous missense variant (NM_005269.3; c.1064C>A; p.(Thr355Asn) in the gene GLI1 segregating with the disease phenotype within the family. This study presents first familial case of autosomal dominant form of polydactyly caused by the GLI1 variant.

摘要

多指畸形是指手足部出现多余的手指/脚趾,且这些多余的手指/脚趾具有或不具有完整的骨结构,是最常见的一种自发性附肢畸形。根据多余的手指/脚趾位于近侧(桡侧)还是远侧(尺侧),可将其分为轴前型多指(PPD)、轴后型多指(PAP)和中央多指(正中多指)。多指畸形至少由 10 个基因引起,这些基因与常染色体显性遗传的多指畸形有关。本研究调查了一个常染色体显性遗传的非综合征性多指畸形家系,通过全外显子测序和 Sanger 测序发现了一个新的杂合错义突变(NM_005269.3; c.1064C>A; p.(Thr355Asn)),该突变位于 GLI1 基因中,与家系中的疾病表型共分离。本研究报道了首例由 GLI1 突变引起的常染色体显性遗传的多指畸形家系。

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