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

被引:0
|
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
来源
关键词
D O I
暂无
中图分类号
学科分类号
摘要
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.
引用
收藏
相关论文
共 50 条
  • [1] 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
    SCIENTIFIC REPORTS, 2017, 7
  • [2] X-linked Ocular Albinism
    Tsang, Stephen H.
    Sharma, Tarun
    ATLAS OF INHERITED RETINAL DISEASES, 2018, 1085 : 49 - 52
  • [3] GENETIC-ANALYSIS IN X-LINKED OCULAR ALBINISM
    DEMARTINVILLE, B
    GRAHAM, JM
    RISDAHL, JS
    RICH, SS
    PEARSON, PL
    MILLER, FS
    KING, RA
    CYTOGENETICS AND CELL GENETICS, 1987, 46 (1-4): : 605 - 606
  • [4] GENETIC-MAPPING OF X-LINKED OCULAR ALBINISM - LINKAGE ANALYSIS IN BRITISH FAMILIES
    CHARLES, SJ
    MOORE, AT
    YATES, JRW
    JOURNAL OF MEDICAL GENETICS, 1992, 29 (08) : 552 - 554
  • [5] Novel GPR143 mutations and clinical characteristics in six Chinese families with X-linked ocular albinism
    Fang, Shaohua
    Guo, Xiangming
    Jia, Xiaoyun
    Xiao, Xueshan
    Li, Shiqiang
    Zhang, Qingjiong
    MOLECULAR VISION, 2008, 14 (230-33): : 1974 - 1982
  • [6] X-LINKED MICROMELANOSOMAL OCULAR ALBINISM
    WITKOP, C
    KING, R
    EFRON, M
    LEBOVITZ, R
    PAULI, R
    YALE JOURNAL OF BIOLOGY AND MEDICINE, 1984, 57 (03): : 433 - 434
  • [7] MACROMELANOSOMES IN X-LINKED OCULAR ALBINISM
    GARNER, A
    JAY, BS
    HISTOPATHOLOGY, 1980, 4 (03) : 243 - 254
  • [8] Identification of two novel mutations in families with X-linked ocular albinism
    Iannaccone, Alessandro
    Gallaher, Kevin T.
    Buchholz, Janda
    Jennings, Barbara J.
    Neitz, Maureen
    Sidjanin, Duska J.
    MOLECULAR VISION, 2007, 13 (204-08): : 1856 - 1861
  • [9] X-LINKED OCULAR ALBINISM IN BLACKS - OCULAR ALBINISM CUM PIGMENTO
    ODONNELL, FE
    GREEN, WR
    FLEISCHMAN, JA
    HAMBRICK, GW
    ARCHIVES OF OPHTHALMOLOGY, 1978, 96 (07) : 1189 - 1192
  • [10] X-LINKED OCULAR ALBINISM - CLINICAL DELINEATION AND LINKAGE ANALYSIS
    GRAHAM, JM
    DEMARTINVILLE, B
    VOLPE, MA
    RISDAHL, JS
    RICH, SS
    PEARSON, PL
    MILLER, FS
    KING, RA
    CLINICAL RESEARCH, 1988, 36 (01): : A207 - A207