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X 连锁眼皮肤白化病 3 个中国家系的分子遗传学及临床评估

Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism.

机构信息

Department of Ophthalmology, Peking Union Medical College Hospital, Peking Union Medical College, Chinese Academy of Medical Sciences, Beijing, China.

出版信息

Sci Rep. 2017 Feb 17;7:33713. doi: 10.1038/srep33713.

Abstract

X-linked ocular albinism (OA1) is an X-linked inherited disease characterized by hypopigmentation of the fundus and nystagmus. Our study performed mutation analysis of the G protein-coupled receptor 143 gene (GPR143) and assessed the clinical characteristics of OA1 in three Chinese families. Three novel mutations, c.333_360+14del42insCTT, c.276G>A (p.W92X), and c.793C>T (p.R265X), were identified in GPR143 by PCR followed by Sanger sequencing in these families. All affected individuals presented with nystagmus, photophobia, poor visual acuity, foveal hypoplasia and varying degrees of hypopigmentation of the fundus. The fundus of female carriers showed pigmented streaks alternating with hypopigmented streaks. These results allowed us to expand the spectrum of mutations in GPR143 and phenotypes associated with ocular albinism.

摘要

X 连锁眼皮肤白化病 1 型(OA1)是一种 X 连锁遗传性疾病,其特征为眼底色素减退和眼球震颤。我们的研究对 G 蛋白偶联受体 143 基因(GPR143)进行了突变分析,并评估了三个中国家系中 OA1 的临床特征。通过 PCR 后 Sanger 测序在这些家系中发现了 GPR143 的三个新突变,c.333_360+14del42insCTT、c.276G>A(p.W92X)和 c.793C>T(p.R265X)。所有受影响的个体均表现为眼球震颤、畏光、视力差、黄斑发育不良和眼底不同程度的色素减退。女性携带者的眼底表现为色素条纹与色素减退条纹交替出现。这些结果扩展了 GPR143 的突变谱和与眼皮肤白化病相关的表型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/84c5/5314354/967011561440/srep33713-f1.jpg

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