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

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 n...

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Autores principales: Zou, Xuan, Li, Hui, Yang, Lizhu, Sun, Zixi, Yuan, Zhisheng, Li, Huajin, Sui, Ruifang
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5314354/
https://www.ncbi.nlm.nih.gov/pubmed/28211458
http://dx.doi.org/10.1038/srep33713
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author Zou, Xuan
Li, Hui
Yang, Lizhu
Sun, Zixi
Yuan, Zhisheng
Li, Huajin
Sui, Ruifang
author_facet Zou, Xuan
Li, Hui
Yang, Lizhu
Sun, Zixi
Yuan, Zhisheng
Li, Huajin
Sui, Ruifang
author_sort Zou, Xuan
collection PubMed
description 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.
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spelling pubmed-53143542017-02-23 Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism Zou, Xuan Li, Hui Yang, Lizhu Sun, Zixi Yuan, Zhisheng Li, Huajin Sui, Ruifang Sci Rep Article 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. Nature Publishing Group 2017-02-17 /pmc/articles/PMC5314354/ /pubmed/28211458 http://dx.doi.org/10.1038/srep33713 Text en Copyright © 2016, The Author(s) http://creativecommons.org/licenses/by/4.0/ This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/
spellingShingle Article
Zou, Xuan
Li, Hui
Yang, Lizhu
Sun, Zixi
Yuan, Zhisheng
Li, Huajin
Sui, Ruifang
Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism
title Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism
title_full Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism
title_fullStr Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism
title_full_unstemmed Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism
title_short Molecular genetic and clinical evaluation of three Chinese families with X-linked ocular albinism
title_sort molecular genetic and clinical evaluation of three chinese families with x-linked ocular albinism
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5314354/
https://www.ncbi.nlm.nih.gov/pubmed/28211458
http://dx.doi.org/10.1038/srep33713
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