• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

以色列遗传性视网膜疾病联合会(IIRDC)对以色列遗传性视网膜疾病进行的全国性基因分析。

A nationwide genetic analysis of inherited retinal diseases in Israel as assessed by the Israeli inherited retinal disease consortium (IIRDC).

机构信息

Department of Ophthalmology, Hadassah Medical Center, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, Israel.

Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel.

出版信息

Hum Mutat. 2020 Jan;41(1):140-149. doi: 10.1002/humu.23903. Epub 2019 Sep 15.

DOI:10.1002/humu.23903
PMID:31456290
Abstract

Inherited retinal diseases (IRDs) cause visual loss due to dysfunction or progressive degeneration of photoreceptors. These diseases show marked phenotypic and genetic heterogeneity. The Israeli IRD consortium (IIRDC) was established in 2013 with the goal of performing clinical and genetic mapping of the majority of Israeli IRD patients. To date, we recruited 2,420 families including 3,413 individuals with IRDs. On the basis of our estimation, these patients represent approximately 40% of Israeli IRD patients. To the best of our knowledge, this is, by far, the largest reported IRD cohort, and one of the first studies addressing the genetic analysis of IRD patients on a nationwide scale. The most common inheritance pattern in our cohort is autosomal recessive (60% of families). The most common retinal phenotype is retinitis pigmentosa (43%), followed by Stargardt disease and cone/cone-rod dystrophy. We identified the cause of disease in 56% of the families. Overall, 605 distinct mutations were identified, of which 12% represent prevalent founder mutations. The most frequently mutated genes were ABCA4, USH2A, FAM161A, CNGA3, and EYS. The results of this study have important implications for molecular diagnosis, genetic screening, and counseling, as well as for the development of new therapeutic strategies for retinal diseases.

摘要

遗传性视网膜疾病(IRDs)由于光感受器的功能障碍或进行性退化而导致视力丧失。这些疾病表现出明显的表型和遗传异质性。以色列 IRD 联盟(IIRDC)成立于 2013 年,目标是对大多数以色列 IRD 患者进行临床和遗传作图。迄今为止,我们已经招募了 2420 个家庭,包括 3413 名 IRD 患者。根据我们的估计,这些患者约占以色列 40%的 IRD 患者。据我们所知,这是迄今为止报道的最大的 IRD 队列之一,也是第一个在全国范围内对 IRD 患者进行遗传分析的研究之一。我们队列中最常见的遗传模式是常染色体隐性遗传(60%的家庭)。最常见的视网膜表型是色素性视网膜炎(43%),其次是 Stargardt 病和锥/锥杆营养不良。我们确定了 56%的家庭的疾病原因。总的来说,我们发现了 605 种不同的突变,其中 12%是常见的突变。最常突变的基因是 ABCA4、USH2A、FAM161A、CNGA3 和 EYS。这项研究的结果对分子诊断、遗传筛查和咨询具有重要意义,也为视网膜疾病的新治疗策略的发展提供了依据。

相似文献

1
A nationwide genetic analysis of inherited retinal diseases in Israel as assessed by the Israeli inherited retinal disease consortium (IIRDC).以色列遗传性视网膜疾病联合会(IIRDC)对以色列遗传性视网膜疾病进行的全国性基因分析。
Hum Mutat. 2020 Jan;41(1):140-149. doi: 10.1002/humu.23903. Epub 2019 Sep 15.
2
[THE ISRAELI INHERITED RETINAL DISEASES CONSORTIUM (IIRDC)- CLINICAL-GENETIC MAPPING AND FUTURE PERSPECTIVES].[以色列遗传性视网膜疾病联盟(IIRDC)——临床-遗传图谱绘制与未来展望]
Harefuah. 2019 Feb;158(2):91-95.
3
Nationwide Prevalence of Inherited Retinal Diseases in the Israeli Population.以色列人群中遗传性视网膜疾病的全国患病率。
JAMA Ophthalmol. 2024 Jul 1;142(7):609-616. doi: 10.1001/jamaophthalmol.2024.1461.
4
Genetic causes of inherited retinal diseases among Israeli Jews of Ethiopian ancestry.遗传性视网膜疾病的遗传原因在埃塞俄比亚裔以色列犹太人中。
Mol Vis. 2023 Apr 22;29:1-12. eCollection 2023.
5
Non-syndromic inherited retinal diseases in Poland: Genes, mutations, and phenotypes.波兰的非综合征遗传性视网膜疾病:基因、突变和表型。
Mol Vis. 2021 Jul 16;27:457-465. eCollection 2021.
6
Novel null mutations in the EYS gene are a frequent cause of autosomal recessive retinitis pigmentosa in the Israeli population.以色列人群中常染色体隐性遗传型视网膜色素变性的一个常见病因是 EYS 基因突变。
Invest Ophthalmol Vis Sci. 2010 Sep;51(9):4387-94. doi: 10.1167/iovs.09-4732. Epub 2010 Apr 7.
7
Genetics of Retinitis Pigmentosa and Other Hereditary Retinal Disorders in Western Switzerland.瑞士西部的色素性视网膜炎和其他遗传性视网膜疾病的遗传学研究
Ophthalmic Res. 2024;67(1):172-182. doi: 10.1159/000536036. Epub 2023 Dec 29.
8
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.
9
Spectrum of Genetic Variants in the Most Common Genes Causing Inherited Retinal Disease in a Large Molecularly Characterized United Kingdom Cohort.在一个经过大规模分子特征分析的英国队列中,最常见的遗传性视网膜疾病相关基因的遗传变异谱。
Ophthalmol Retina. 2024 Jul;8(7):699-709. doi: 10.1016/j.oret.2024.01.012. Epub 2024 Jan 12.
10
Identification of mutations causing inherited retinal degenerations in the israeli and palestinian populations using homozygosity mapping.利用纯合子作图技术鉴定导致以色列和巴勒斯坦人群遗传性视网膜变性的突变。
Invest Ophthalmol Vis Sci. 2014 Feb 24;55(2):1149-60. doi: 10.1167/iovs.13-13625.

引用本文的文献

1
Phenotypic and Genotypic Characterization of 171 Patients with Syndromic Inherited Retinal Diseases Highlights the Importance of Genetic Testing for Accurate Clinical Diagnosis.171例综合征性遗传性视网膜疾病患者的表型和基因型特征凸显了基因检测对准确临床诊断的重要性。
Genes (Basel). 2025 Jun 26;16(7):745. doi: 10.3390/genes16070745.
2
RetiGene, a comprehensive gene atlas for inherited retinal diseases (IRDs).RetiGene,一种用于遗传性视网膜疾病(IRD)的综合基因图谱。
bioRxiv. 2025 Jun 8:2025.06.08.653722. doi: 10.1101/2025.06.08.653722.
3
ABCA4-associated disease in childhood and adolescence- a phenotype study.
儿童及青少年ABCA4相关疾病——一项表型研究
Graefes Arch Clin Exp Ophthalmol. 2025 Jul 11. doi: 10.1007/s00417-025-06884-9.
4
AAV2.7m8 transduction of stage 2 human retinal organoids induces highly variable responses in innate and inflammatory gene expression and cytokine secretion.2型腺相关病毒(AAV2.7m8)转导2期人类视网膜类器官会在先天性和炎症基因表达以及细胞因子分泌方面引发高度可变的反应。
Exp Eye Res. 2025 Jun 6;258:110478. doi: 10.1016/j.exer.2025.110478.
5
A novel founder variant in BEST1 gene causing autosomal recessive bestrophinopathy.BEST1基因中的一种新型奠基者变异导致常染色体隐性遗传性贝斯特病。
Orphanet J Rare Dis. 2025 May 25;20(1):248. doi: 10.1186/s13023-025-03813-1.
6
Clinical Exome-Based Redefinition and Reclassification of Retinitis Pigmentosa.基于临床外显子组的视网膜色素变性重新定义与重新分类
J Korean Med Sci. 2025 Apr 28;40(16):e54. doi: 10.3346/jkms.2025.40.e54.
7
A comprehensive genetic landscape of inherited retinal diseases in a large Pakistani cohort.一个大型巴基斯坦队列中遗传性视网膜疾病的综合遗传图谱。
NPJ Genom Med. 2025 Apr 4;10(1):31. doi: 10.1038/s41525-025-00488-2.
8
The importance of genetic counselling and testing in inherited eye diseases: A population-based retrospective study.遗传性眼病中遗传咨询与检测的重要性:一项基于人群的回顾性研究。
PLoS One. 2025 Feb 13;20(2):e0318492. doi: 10.1371/journal.pone.0318492. eCollection 2025.
9
Clinical and genetic landscape of IRD in Portugal: pooled data from the nationwide IRD-PT registry.葡萄牙IRD的临床和遗传概况:来自全国IRD-PT登记处的汇总数据。
NPJ Genom Med. 2025 Feb 12;10(1):11. doi: 10.1038/s41525-025-00475-7.
10
Characterizing the Genetic Basis for Inherited Retinal Disease: Lessons Learned From the Foundation Fighting Blindness Clinical Consortium's Gene Poll.遗传性视网膜疾病遗传基础的特征分析:从抗盲基金会临床联盟基因调查中汲取的经验教训。
Invest Ophthalmol Vis Sci. 2025 Feb 3;66(2):12. doi: 10.1167/iovs.66.2.12.