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日本人群的眼部遗传学。

Ocular genetics in the Japanese population.

机构信息

Hamamatsu University School of Medicine, 1-20-1 Handayama, Chuo-ku, Hamamatsu city, Shizuoka, 431-3192, Japan.

出版信息

Jpn J Ophthalmol. 2024 Sep;68(5):401-418. doi: 10.1007/s10384-024-01109-8. Epub 2024 Sep 14.

DOI:10.1007/s10384-024-01109-8
PMID:39271608
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11420330/
Abstract

In today's globalized society, ophthalmologists can examine people of different ethnicities regardless of where they live. The frequency of disease-causing genes varies according to a patient's ethnic background. We explain genetic findings for Japanese patients with inherited eye diseases. Ocular genetics has made great advances over the past 30 years. For example, detecting mutations at nucleotide position 11778 in mitochondrial DNA was useful in the genetic diagnosis of Leber's hereditary optic neuropathy (LHON). I evaluated the genotype-phenotype relationship in cases of corneal dystrophy and inherited retinal dystrophy (IRD). I identified the entire exon sequence of the eyes shut homolog (EYS) gene in patients with autosomal recessive retinitis pigmentosa (RP). EYS gene mutations are the most frequent cause of autosomal recessive RP. RPGRIP1 may be a common causative gene with early-onset severe retinal dystrophy, including Leber congenital amaurosis. However, some genes have complex structures that are difficult to analyze, including the OPN1LW/OPN1MW gene cluster in blue cone monochromacy and the IKBKG/NEMO genes in incontinentia pigmenti. This review will also present two cases with uniparental disomy, a case of IRD with double mutations, and a case with RP complicated with LHON-like neuropathy. Precise understanding of the effects of genetic variants may reveal differences in the clinical characteristics of patients with the same variant. When starting genome medicine, accurately diagnosing the patient, making accurate prediction, determining the genetic pattern, and providing genetic counseling are important. Above all, that both the doctors and patients understand genetic diseases correctly is important.

摘要

在当今全球化的社会中,眼科医生可以为不同种族的人进行检查,无论他们居住在哪里。致病基因的频率因患者的种族背景而异。我们对日本遗传性眼病患者的遗传发现进行了解释。眼科学在过去 30 年中取得了巨大的进展。例如,检测线粒体 DNA 核苷酸位置 11778 的突变在莱伯遗传性视神经病变(LHON)的遗传诊断中非常有用。我评估了角膜营养不良和遗传性视网膜病变(IRD)的基因型-表型关系。我在常染色体隐性视网膜色素变性(RP)患者中鉴定了眼睛关闭同源物(EYS)基因的整个外显子序列。EYS 基因突变是常染色体隐性 RP 的最常见原因。RPGRIP1 可能是一种常见的致病基因,与包括莱伯先天性黑矇在内的早发性严重视网膜营养不良有关。然而,一些基因具有复杂的结构,难以分析,包括蓝色锥体单色性中的 OPN1LW/OPN1MW 基因簇和 incontinentia pigmenti 中的 IKBKG/NEMO 基因。本综述还将介绍两例单亲二体性病例、一例具有双重突变的 IRD 病例和一例伴有 LHON 样神经病的 RP 病例。对遗传变异影响的精确理解可能揭示出具有相同变异的患者在临床特征上的差异。当启动基因组医学时,准确诊断患者、进行准确预测、确定遗传模式和提供遗传咨询非常重要。最重要的是,医生和患者都要正确理解遗传疾病。

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本文引用的文献

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Gene Variants Are a Frequent Cause of a Rare Disease: Leber Hereditary Optic Neuropathy in Polish Patients.基因突变是一种罕见疾病的常见病因:波兰患者的莱伯遗传性视神经病变。
Int J Mol Sci. 2023 Dec 15;24(24):17496. doi: 10.3390/ijms242417496.
2
The Structural Abnormalities Are Deeply Involved in the Cause of -Related Retinal Dystrophy in Japanese Patients.结构异常与日本患者的 -相关视网膜营养不良的病因密切相关。
Int J Mol Sci. 2023 Sep 5;24(18):13678. doi: 10.3390/ijms241813678.
3
Characterizing the genotypic spectrum of retinitis pigmentosa in East Asian populations: a systematic review.
中文译文:系统综述东亚人群中色素性视网膜炎的基因型谱。
Ophthalmic Genet. 2023 Apr;44(2):109-118. doi: 10.1080/13816810.2023.2182329. Epub 2023 Mar 1.
4
Genotype and phenotype characteristics of RHO-associated retinitis pigmentosa in the Japanese population.日本人群中 Rho 相关视网膜炎色素变性的基因型和表型特征。
Jpn J Ophthalmol. 2023 Mar;67(2):138-148. doi: 10.1007/s10384-023-00975-y. Epub 2023 Jan 17.
5
Genetic characterization of 1210 Japanese pedigrees with inherited retinal diseases by whole-exome sequencing.通过全外显子组测序对1210个患有遗传性视网膜疾病的日本家系进行基因特征分析。
Hum Mutat. 2022 Dec;43(12):2251-2264. doi: 10.1002/humu.24492. Epub 2022 Nov 7.
6
The landscape of submicroscopic structural variants at the gene cluster on Xq28 underlying blue cone monochromacy.Xq28 基因簇上导致蓝色锥细胞单色视的亚微观结构变异景观。
Proc Natl Acad Sci U S A. 2022 Jul 5;119(27):e2115538119. doi: 10.1073/pnas.2115538119. Epub 2022 Jun 27.
7
Likely pathogenic structural variants in genetically unsolved patients with retinitis pigmentosa revealed by long-read sequencing.长读测序揭示遗传性视网膜色素变性未解决患者中可能致病的结构变异。
J Med Genet. 2022 Nov;59(11):1133-1138. doi: 10.1136/jmedgenet-2022-108428. Epub 2022 Jun 15.
8
Maternal Uniparental Isodisomy of Chromosome 4 and 8 in Patients with Retinal Dystrophy: -Congenital Disorders of Glycosylation and -Related Retinitis Pigmentosa.视网膜营养不良患者中4号和8号染色体的母源单亲二体:糖基化先天性疾病及相关色素性视网膜炎
Genes (Basel). 2022 Feb 16;13(2):359. doi: 10.3390/genes13020359.
9
Retinitis pigmentosa with optic neuropathy and mutations: A case report.伴有视神经病变及突变的色素性视网膜炎:一例报告
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Genotype-Phenotype Correlations in -Associated Retinal Dystrophies: A Multi-Center Cohort Study in JAPAN.与视网膜营养不良相关的基因型-表型相关性:日本的一项多中心队列研究
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