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

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作者
Xuan Zou
Hui Li
Lizhu Yang
Zixi Sun
Zhisheng Yuan
Huajin Li
Ruifang Sui
机构
[1] Peking Union Medical College Hospital,Department of Ophthalmology
[2] Peking Union Medical College,undefined
[3] Chinese Academy of Medical Sciences,undefined
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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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