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从白内障到综合征诊断:1 型沃伯格微综合征患者的再评估。

From cataract to syndrome diagnosis: Revaluation of Warburg-Micro syndrome Type 1 patients.

机构信息

Department of Pediatric Genetics, Ankara Bilkent City Hospital, Ankara, Turkey.

Department of Pediatric Genetics, Marmara University, İstanbul, Turkey.

出版信息

Am J Med Genet A. 2021 Aug;185(8):2325-2334. doi: 10.1002/ajmg.a.62234. Epub 2021 May 5.

Abstract

Warburg-Micro syndrome (WARBM) is a rare autosomal recessively inherited neuro-ophthalmologic syndrome. Although WARBM shows genetic heterogeneity, the pathogenic variants in RAB3GAP1 were the most common cause of WARBM. In this study, we aimed to evaluate the detailed clinical and dysmorphic features of seven WARBM1 patients and overview the variant spectrum of RAB3GAP1 in comparison with the literature who were referred due to congenital cataracts. A previously reported homozygous variant (c.2187_2188delGAinsCT) was identified in three of these patients, while the other four had three novel variants (c.251_258delAGAA, c.2606+1G>A, and c.2861_2862dupGC). Congenital cataract and corpus callosum hypo/agenesia are pathognomonic for WARBM, which could be distinguished from other similar syndromes with additional typical dysmorphic facial features. Although there is no known phenotype and genotype correlation in any type of WARBM, RAB3GAP1 gene analysis should be previously requested as the first step of genetic diagnosis in clinically suspicious patients when it is not possible to request a multi-gene panel.

摘要

沃伯格-微综合征(WARBM)是一种罕见的常染色体隐性遗传的神经眼科学综合征。尽管 WARBM 表现出遗传异质性,但 RAB3GAP1 的致病变异是 WARBM 最常见的原因。在这项研究中,我们旨在评估 7 名 WARBM1 患者的详细临床和发育不良特征,并与因先天性白内障而就诊的文献中 RAB3GAP1 的变异谱进行综述。在这 3 名患者中发现了先前报道的纯合变异(c.2187_2188delGAinsCT),而其他 4 名患者有 3 种新的变异(c.251_258delAGAA、c.2606+1G>A 和 c.2861_2862dupGC)。先天性白内障和胼胝体发育不全/发育不良是 WARBM 的特征性表现,这可以与其他具有附加典型发育不良面部特征的相似综合征区分开来。尽管在任何类型的 WARBM 中都没有已知的表型和基因型相关性,但当无法请求多基因组时,应在临床可疑患者中作为遗传诊断的第一步预先请求 RAB3GAP1 基因分析。

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