Literature DB >> 28112372

Identification of a missense mutation in the tyrosinase gene in a Chinese family with oculocutaneous albinism type 1.

Qian Lu1, Lamei Yuan1, Hongbo Xu1, Xiangjun Huang1, Zhijian Yang1, Junhui Yi2, Bin Ni3, Yong Chen3, Hao Deng1.   

Abstract

Oculocutaneous albinism (OCA) is a group of heterogeneous and autosomal recessive disorders characterized by a reduction or complete loss of melanin biosynthesis in melanocytes. OCA type 1 (OCA1) is the most severe and common form of OCA, and is caused by mutations in the tyrosinase gene (TYR). The present study aimed to identify the genetic cause of OCA1 in a four‑generation consanguineous Chinese Han family. Complete physical examinations were performed and blood samples were collected from five members of the family and 100 unrelated healthy controls. Exome sequencing was conducted in the proband, followed by verification in other family members, using Sanger sequencing. Patients in the family presented with typical OCA1 features, including hypopigmentation of the skin and hair, and distinctive ocular changes. A homozygous missense variant, c.896G>A (p.R299H), in the TYR gene was identified in two patients, which co‑segregated with disease in the family. This variant was not present in the 100 healthy controls. These results expand the number of mutations identified to be responsible for OCA1 in the Chinese Han population, and may have implications for genetic counseling and clinical management of the disease.

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Year:  2017        PMID: 28112372     DOI: 10.3892/mmr.2017.6137

Source DB:  PubMed          Journal:  Mol Med Rep        ISSN: 1791-2997            Impact factor:   2.952


  2 in total

1.  Novel and Recurring Disease-Causing NF1 Variants in Two Chinese Families with Neurofibromatosis Type 1.

Authors:  Heng Xiao; Lamei Yuan; Hongbo Xu; Zhijian Yang; Feizhou Huang; Zhi Song; Yan Yang; Cheng Zeng; Hao Deng
Journal:  J Mol Neurosci       Date:  2018-07-25       Impact factor: 3.444

2.  Identification of a GNE homozygous mutation in a Han-Chinese family with GNE myopathy.

Authors:  Yuan Wu; Lamei Yuan; Yi Guo; Anjie Lu; Wen Zheng; Hongbo Xu; Yan Yang; Pengzhi Hu; Shaojuan Gu; Bingqi Wang; Hao Deng
Journal:  J Cell Mol Med       Date:  2018-08-29       Impact factor: 5.310

  2 in total

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