Department of Biochemistry, Quaid-i-Azam University, Islamabad, Pakistan; Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Exp Eye Res. 2024 Jul;244:109945. doi: 10.1016/j.exer.2024.109945. Epub 2024 May 28.
Inherited retinal dystrophies (IRDs) are characterized by photoreceptor dysfunction or degeneration. Clinical and phenotypic overlap between IRDs makes the genetic diagnosis very challenging and comprehensive genomic approaches for accurate diagnosis are frequently required. While there are previous studies on IRDs in Pakistan, causative genes and variants are still unknown for a significant portion of patients. Therefore, there is a need to expand the knowledge of the genetic spectrum of IRDs in Pakistan. Here, we recruited 52 affected and 53 normal individuals from 15 consanguineous Pakistani families presenting non-syndromic and syndromic forms of IRDs. We employed single molecule Molecular Inversion Probes (smMIPs) based panel sequencing and whole genome sequencing to identify the probable disease-causing variants in these families. Using this approach, we obtained a 93% genetic solve rate and identified 16 (likely) causative variants in 14 families, of which seven novel variants were identified in ATOH7, COL18A1, MERTK, NDP, PROM1, PRPF8 and USH2A while nine recurrent variants were identified in CNGA3, CNGB1, HGSNAT, NMNAT1, SIX6 and TULP1. The novel MERTK variant and one recurrent TULP1 variant explained the intra-familial locus heterogeneity in one of the screened families while two recurrent CNGA3 variants explained compound heterozygosity in another family. The identification of variants in known disease-associated genes emphasizes the utilization of time and cost-effective screening approaches for rapid diagnosis. The timely genetic diagnosis will not only identify any associated systemic issues in case of syndromic IRDs, but will also aid in the acceleration of personalized medicine for patients affected with IRDs.
遗传性视网膜病变(IRDs)的特征是感光细胞功能障碍或变性。IRDs 的临床和表型重叠使得基因诊断极具挑战性,因此经常需要采用全面的基因组方法进行准确诊断。虽然以前在巴基斯坦开展过 IRD 相关研究,但仍有很大一部分患者的致病基因和变异尚不清楚。因此,有必要扩大对巴基斯坦 IRD 遗传谱的认识。在这里,我们招募了 15 个有血缘关系的巴基斯坦家庭的 52 名受影响者和 53 名正常个体,这些家庭呈现出非综合征和综合征形式的 IRD。我们采用单分子分子反转探针(smMIPs)基于面板测序和全基因组测序来识别这些家庭中可能的致病变异。通过这种方法,我们获得了 93%的遗传解决率,并在 14 个家庭中确定了 16 个(可能)致病变异,其中 7 个新变异在 ATOH7、COL18A1、MERTK、NDP、PROM1、PRPF8 和 USH2A 中发现,9 个重复变异在 CNGA3、CNGB1、HGSNAT、NMNAT1、SIX6 和 TULP1 中发现。新的 MERTK 变异和一个重复的 TULP1 变异解释了一个筛查家庭中的家族内基因座异质性,而两个重复的 CNGA3 变异解释了另一个家庭的复合杂合性。已知疾病相关基因中的变异的鉴定强调了利用时间和具有成本效益的筛查方法进行快速诊断的重要性。及时的基因诊断不仅可以识别综合征性 IRD 中的任何相关系统性问题,还可以帮助加速患有 IRD 的患者的个性化医疗。