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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.

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。

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