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卡塔尔非综合征性听力损失相关基因检测的遗传基础和诊断收益。

The genetic basis and the diagnostic yield of genetic testing related to nonsyndromic hearing loss in Qatar.

机构信息

Department of Biomedical Sciences, College of Health Sciences, QU Health, Qatar University, Doha, Qatar.

Department of Adult and Pediatric Medical Genetics, Hamad Medical Corporation, Doha, Qatar.

出版信息

Sci Rep. 2024 Feb 20;14(1):4202. doi: 10.1038/s41598-024-52784-z.

Abstract

Hearing loss is the most predominant sensory defect occurring in pediatrics, of which, 66% cases are attributed to genetic factors. The prevalence of hereditary hearing loss increases in consanguineous populations, and the prevalence of hearing loss in Qatar is 5.2%. We aimed to investigate the genetic basis of nonsyndromic hearing loss (NSHL) in Qatar and to evaluate the diagnostic yield of different genetic tests available. A retrospective chart review was conducted for 59 pediatric patients with NSHL referred to the Department of Adult and Pediatric Medical Genetics at Hamad Medical Corporation in Qatar, and who underwent at least one genetic test. Out of the 59 patients, 39 were solved cases due to 19 variants in 11 genes and two copy number variants that explained the NSHL phenotype. Of them 2 cases were initially uncertain and were reclassified using familial segregation. Around 36.8% of the single variants were in GJB2 gene and c.35delG was the most common recurrent variant seen in solved cases. We detected the c.283C > T variant in FGF3 that was seen in a Qatari patient and found to be associated with NSHL for the first time. The overall diagnostic yield was 30.7%, and the diagnostic yield was significantly associated with genetic testing using GJB2 sequencing and using the hearing loss (HL) gene panel. The diagnostic yield for targeted familial testing was 60% (n = 3 patients) and for gene panel was 50% (n = 5). Thus, we recommend using GJB2 gene sequencing as a first-tier genetic test and HL gene panel as a second-tier genetic test for NSHL. Our work provided new insights into the genetic pool of NSHL among Arabs and highlights its unique diversity, this is believed to help further in the diagnostic and management options for NSHL Arab patients.

摘要

听力损失是儿科最常见的感觉缺陷,其中 66%的病例归因于遗传因素。遗传性听力损失在近亲人群中的发病率增加,卡塔尔的听力损失发病率为 5.2%。我们旨在研究卡塔尔非综合征性听力损失(NSHL)的遗传基础,并评估可用的不同遗传检测的诊断效果。我们对 59 名被转诊至卡塔尔哈马德医疗保健公司成人和儿科医学遗传学系的 NSHL 儿科患者进行了回顾性图表审查,这些患者至少接受了一项遗传检测。在 59 名患者中,由于 11 个基因中的 19 个变体和 2 个拷贝数变异导致 39 名患者为已解决病例,这些变体解释了 NSHL 表型。其中 2 例最初不确定,使用家族分离重新分类。大约 36.8%的单变体位于 GJB2 基因中,c.35delG 是已解决病例中最常见的反复出现变体。我们在一名卡塔尔患者中检测到 FGF3 中的 c.283C > T 变体,这是首次发现与 NSHL 相关。总体诊断率为 30.7%,使用 GJB2 测序和听力损失(HL)基因谱进行遗传检测与诊断率显著相关。针对家族性检测的诊断率为 60%(n = 3 例),针对基因谱的诊断率为 50%(n = 5 例)。因此,我们建议将 GJB2 基因测序作为 NSHL 的一线遗传检测,HL 基因谱作为二线遗传检测。我们的工作为阿拉伯人中 NSHL 的遗传库提供了新的见解,并强调了其独特的多样性,这有助于进一步为阿拉伯 NSHL 患者提供诊断和管理选择。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6203/10879212/51b099cee13d/41598_2024_52784_Fig1_HTML.jpg

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