Department of Zoology, Faculty of Biological Sciences, Quaid-i-Azam University, University Road, Islamabad 45320, Pakistan.
Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA.
Genes (Basel). 2022 Sep 10;13(9):1630. doi: 10.3390/genes13091630.
Inherited retinal dystrophies (IRDs) are a heterogeneous group of degenerative disorders of the retina. Retinitis Pigmentosa (RP) is a common type of IRD that causes night blindness and loss of peripheral vision and may progress to blindness. Mutations in more than 300 genes have been associated with syndromic and non-syndromic IRDs. Recessive forms are more frequent in populations where endogamy is a social preference, such as Pakistan. The aim of this study was to identify molecular determinants of IRDs with the common presentation of night blindness in consanguineous Pakistani families. This study included nine consanguineous IRD-affected families that presented autosomal recessive inheritance of the night blindness phenotype. DNA was extracted from blood samples. Targeted exome sequencing of 344 known genes for retinal dystrophies was performed. Screening of nine affected families revealed two novel (c.5571_5576delinsCTAGATand c.471dup in EYS and SPATA7 genes, respectively) and six reported pathogenic mutations (c.304C>A, c.187C>T, c.1560C>A, c.547C>T, c.109del and c.9911_11550del in PDE6A, USH2A, USH2A, NMNAT1, PAX6 and ALMS1 genes, respectively) segregating with disease phenotype in each respective family. Molecular determinants of hereditary retinal dystrophies were identified in all screened families. Identification of novel variants aid future diagnosis of retinal dystrophies and help to provide genetic counseling to affected families.
遗传性视网膜病变(IRDs)是一组异质性的视网膜退行性疾病。色素性视网膜炎(RP)是一种常见的 IRD,可导致夜盲症和周边视力丧失,并可能发展为失明。超过 300 个基因的突变与综合征性和非综合征性 IRD 有关。在像巴基斯坦这样的内婚制是一种社会偏好的人群中,隐性形式更为常见。本研究的目的是确定具有夜盲症共同表现的 IRD 的分子决定因素,这些患者来自有血缘关系的巴基斯坦家庭。本研究包括 9 个有血缘关系的 IRD 受累家庭,这些家庭表现出自体隐性遗传的夜盲症表型。从血液样本中提取 DNA。对 344 个已知的视网膜病变基因进行靶向外显子组测序。对 9 个受影响的家庭进行筛查,发现了两个新的(c.5571_5576delinsCTAGAT 和 c.471dup,分别位于 EYS 和 SPATA7 基因中)和 6 个报道的致病性突变(c.304C>A、c.187C>T、c.1560C>A、c.547C>T、c.109del 和 c.9911_11550del,分别位于 PDE6A、USH2A、USH2A、NMNAT1、PAX6 和 ALMS1 基因中),在每个家族中与疾病表型共分离。在所筛选的家庭中均确定了遗传性视网膜病变的分子决定因素。新型变异体的鉴定有助于未来视网膜病变的诊断,并有助于为受影响的家庭提供遗传咨询。