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ANKRD24 中的一个移码变异表明其在人类非综合征性听力损失中的作用。

A Frameshift Variant in ANKRD24 Implicates Its Role in Human Non-Syndromic Hearing Loss.

作者信息

Kazemi Negar, Rezvani Rezvandeh Raziye, Zare Ashrafi Farzane, Shokouhian Ebrahim, Edizadeh Masoud, Booth Kevin T A, Kahrizi Kimia, Najmabadi Hossein, Mohseni Marzieh

机构信息

Genetics Research Center, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran.

Department of Bioinformatics, Genoks Genetic Diagnosis Center, Ankara, Turkey.

出版信息

Clin Genet. 2025 Feb;107(2):214-218. doi: 10.1111/cge.14635. Epub 2024 Oct 21.

DOI:10.1111/cge.14635
PMID:39434538
Abstract

Hearing loss (HL) is the most prevalent sensorineural disorders, affecting about one in 1000 newborns. Over half of the cases are attributed to genetic factors; however, due to the extensive clinical and genetic heterogeneity, many cases remain without a conclusive genetic diagnosis. The advent of next-generation sequencing methodologies in recent years has greatly helped unravel the genetic etiology of HL by identifying numerous genes and causative variants. Despite this, much remains to be uncovered about the genetic basis of sensorineural hearing loss (SNHL). Here, we report an Iranian consanguineous family with postlingual, moderate-to-severe autosomal recessive SNHL. After first excluding plausible variants in known deafness-causing genes using whole exome sequencing, we reanalyzed the data, using a gene/variant prioritization pipeline established for novel gene discovery for HL. This approach identified a novel homozygous frameshift variant c.1934_1937del; (p.Thr645Lysfs*52) in ANKRD24, which segregated with the HL phenotype in the family. Recently, ANKRD24 has been shown to be a pivotal constituent of the stereocilia rootlet in cochlea hair cells and interacts with TRIOBP, a protein already implicated in human deafness. Our data implicate for the first time, ANKRD24 in human nonsyndromic HL (NSHL) and expands the genetic spectrum of HL.

摘要

听力损失(HL)是最常见的感音神经性疾病,每1000名新生儿中约有1人受其影响。超过一半的病例归因于遗传因素;然而,由于广泛的临床和遗传异质性,许多病例仍未得到明确的基因诊断。近年来,新一代测序方法的出现极大地有助于通过识别众多基因和致病变异来揭示HL的遗传病因。尽管如此,关于感音神经性听力损失(SNHL)的遗传基础仍有许多有待发现。在这里,我们报告了一个伊朗近亲家庭,该家庭患有语后中度至重度常染色体隐性SNHL。首先使用全外显子组测序排除已知致聋基因中可能的变异后,我们使用为HL新基因发现建立的基因/变异优先级排序流程重新分析了数据。这种方法在ANKRD24中鉴定出一种新的纯合移码变异c.1934_1937del;(p.Thr645Lysfs*52),该变异与该家族中的HL表型共分离。最近,ANKRD24已被证明是耳蜗毛细胞静纤毛小根的关键组成部分,并与TRIOBP相互作用,TRIOBP是一种已与人类耳聋有关的蛋白质。我们的数据首次表明ANKRD24与人类非综合征性HL(NSHL)有关,并扩展了HL的遗传谱。

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