Literature DB >> 11449318

Screening for CRX gene mutations in Chinese patients with Leber congenital amaurosis and mutational phenotype.

Q Zhang1, S Li, X Guo, L Guo, X Xiao, X Jia, Z Kuang.   

Abstract

PURPOSE: To screen for possible disease-causing mutations in the CRX gene in Chinese patients with Leber congenital amaurosis (LCA) and to enrich the understanding of its mutational phenotype.
METHODS: Genomic DNA was collected from 27 patients with LCA. The coding sequences of the CRX gene were analyzed using the PCR-heteroduplex-SSCP method. Mutations were confirmed by DNA sequencing.
RESULTS: We identified two heterozygous variations in the CRX gene in two patients with LCA. One was a deletion (GCC-->-CC, A181D1bp) leading to a frameshift and protein truncation. This mutation was present in a patient with LCA, but not in his healthy parents. The ocular manifestations of this A181Delta1bp mutation are described. An intronic variation (IVS1-13G-->C) was found in a patient with LCA as well as in his healthy father.
CONCLUSION: A heterozygous A181D1bp mutation in the CRX gene caused an LCA phenotype in a Chinese patient.

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Year:  2001        PMID: 11449318     DOI: 10.1076/opge.22.2.89.2227

Source DB:  PubMed          Journal:  Ophthalmic Genet        ISSN: 1381-6810            Impact factor:   1.803


  6 in total

1.  Detection of variants in 15 genes in 87 unrelated Chinese patients with Leber congenital amaurosis.

Authors:  Lin Li; Xueshan Xiao; Shiqiang Li; Xiaoyun Jia; Panfeng Wang; Xiangming Guo; Xiaodong Jiao; Qingjiong Zhang; J Fielding Hejtmancik
Journal:  PLoS One       Date:  2011-05-13       Impact factor: 3.240

2.  Mutations of 60 known causative genes in 157 families with retinitis pigmentosa based on exome sequencing.

Authors:  Yan Xu; Liping Guan; Tao Shen; Jianguo Zhang; Xueshan Xiao; Hui Jiang; Shiqiang Li; Jianhua Yang; Xiaoyun Jia; Ye Yin; Xiangming Guo; Jun Wang; Qingjiong Zhang
Journal:  Hum Genet       Date:  2014-06-18       Impact factor: 4.132

Review 3.  Mechanisms of blindness: animal models provide insight into distinct CRX-associated retinopathies.

Authors:  Nicholas M Tran; Shiming Chen
Journal:  Dev Dyn       Date:  2014-06-27       Impact factor: 3.780

4.  Low prevalence of lecithin retinol acyltransferase mutations in patients with Leber congenital amaurosis and autosomal recessive retinitis pigmentosa.

Authors:  Meredith O Sweeney; Terri L McGee; Eliot L Berson; Thaddeus P Dryja
Journal:  Mol Vis       Date:  2007-04-05       Impact factor: 2.367

5.  Mechanistically distinct mouse models for CRX-associated retinopathy.

Authors:  Nicholas M Tran; Alan Zhang; Xiaodong Zhang; Julie B Huecker; Anne K Hennig; Shiming Chen
Journal:  PLoS Genet       Date:  2014-02-06       Impact factor: 5.917

6.  CrxRdy Cat: A Large Animal Model for CRX-Associated Leber Congenital Amaurosis.

Authors:  Laurence M Occelli; Nicholas M Tran; Kristina Narfström; Shiming Chen; Simon M Petersen-Jones
Journal:  Invest Ophthalmol Vis Sci       Date:  2016-07-01       Impact factor: 4.799

  6 in total

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