Lu Qian, Yuan Lamei, Xu Hongbo, Huang Xiangjun, Yang Zhijian, Yi Junhui, Ni Bin, Chen Yong, Deng Hao
Center for Experimental Medicine, The Third Xiangya Hospital, Central South University, Changsha, Hunan 410013, P.R. China.
Department of Ophthalmology, The Third Xiangya Hospital, Central South University, Changsha, Hunan 410013, P.R. China.
Mol Med Rep. 2017 Mar;15(3):1426-1430. doi: 10.3892/mmr.2017.6137. Epub 2017 Jan 23.
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.
眼皮肤白化病(OCA)是一组异质性常染色体隐性疾病,其特征是黑素细胞中黑色素生物合成减少或完全丧失。1型眼皮肤白化病(OCA1)是OCA最严重且最常见的形式,由酪氨酸酶基因(TYR)突变引起。本研究旨在确定一个四代近亲通婚的中国汉族家庭中OCA1的遗传病因。对该家庭的五名成员和100名无关健康对照进行了全面体格检查并采集了血样。先证者进行了外显子组测序,随后使用桑格测序法对其他家庭成员进行验证。该家庭中的患者表现出典型的OCA1特征,包括皮肤和头发色素减退以及独特的眼部变化。在两名患者中鉴定出TYR基因中的纯合错义变体c.896G>A(p.R299H),该变体在家族中与疾病共分离。在100名健康对照中未发现该变体。这些结果扩大了在中国汉族人群中鉴定出的导致OCA1的突变数量,可能对该疾病的遗传咨询和临床管理具有意义。