Department of Biochemistry School of Medicine Ewha Womans University Seoul, Korea.
Exp Mol Med. 2011 Jul 30;43(7):393-400. doi: 10.3858/emm.2011.43.7.043.
Granular corneal dystrophy, type II (CGD2; Avellino corneal dystrophy) is the most common corneal dystrophy among Koreans, but its pathophysiology is still poorly understood. Many reports showed that even though the causative mutation is the same TGFBI R124H mutation, there are severe and mild phenotypes of the corneal dystrophy. We also observed the phenotype differences in our samples. For this reason, we focused our effort on the identification of unknown genetic factor related to phenotype variation. A total 551 individuals from 59 families were genotyped with SNP chip and used in genome-wide linkage analysis. From single-point linkage analyses, we confirmed the known 5q31 region for TGFBI gene, and selected novel nine candidate loci for CGD2. In simulation analysis, the only 3q26.3 region including neuroligin 1 gene (NLGN1) was supported by empirical statistic significance. To investigate the effect of genetic heterogeneity in linkage analysis, we classified CGD2 families into two subgroups. Although we could not find a significant evidence for correlation between the 3q26.3 region and CGD2 phenotypes, this first genome-wide analysis with CGD2 families in Korea has a very important value for offering insights in genetics of CGD2. In addition, the co-segregating loci with CGD2 including 3q26.3 would be a good target for further study to understand the pathophysiology of CGD2.
颗粒状角膜营养不良,II 型(CGD2;阿韦利诺角膜营养不良)是韩国最常见的角膜营养不良,但它的病理生理学仍知之甚少。许多报告表明,即使致病突变是相同的 TGFBI R124H 突变,角膜营养不良也有严重和轻度的表型。我们在样本中也观察到了表型差异。出于这个原因,我们专注于鉴定与表型变异相关的未知遗传因素。我们使用 SNP 芯片对来自 59 个家庭的 551 个人进行了基因分型,并进行了全基因组连锁分析。通过单点连锁分析,我们证实了 TGFBI 基因的已知 5q31 区域,并为 CGD2 选择了 9 个新的候选位点。在模拟分析中,仅包括神经黏附素 1 基因(NLGN1)的 3q26.3 区域得到了经验统计意义的支持。为了研究连锁分析中遗传异质性的影响,我们将 CGD2 家系分为两个亚组。尽管我们没有发现 3q26.3 区域与 CGD2 表型之间存在相关性的显著证据,但这项针对韩国 CGD2 家系的全基因组分析具有非常重要的价值,可以深入了解 CGD2 的遗传学。此外,与 CGD2 共分离的包括 3q26.3 在内的位点将是进一步研究理解 CGD2 病理生理学的良好目标。