ANKRD24 中的一个帧移位变异暗示了它在人类非突发性听力损失中的作用。

IF 2.9 3区 医学 Q2 GENETICS & HEREDITY Clinical Genetics Pub Date : 2024-10-21 DOI:10.1111/cge.14635
Negar Kazemi, Raziye Rezvani Rezvandeh, Farzane Zare Ashrafi, Ebrahim Shokouhian, Masoud Edizadeh, Kevin T A Booth, Kimia Kahrizi, Hossein Najmabadi, Marzieh Mohseni
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引用次数: 0

摘要

听力损失(HL)是最常见的感音神经性疾病,大约每 1000 个新生儿中就有一个听力损失患者。半数以上的病例可归因于遗传因素;然而,由于临床和遗传的广泛异质性,许多病例仍无法得到确凿的遗传诊断。近年来,下一代测序方法的出现通过鉴定大量基因和致病变体,极大地帮助揭示了 HL 的遗传病因。尽管如此,感音神经性听力损失(SNHL)的遗传基础仍有许多问题有待揭示。在此,我们报告了一个患有舌后中重度常染色体隐性 SNHL 的伊朗近亲家庭。我们首先利用全外显子测序排除了已知致聋基因中可能存在的变异,然后利用为发现 HL 新基因而建立的基因/变异优先级管道重新分析了数据。这种方法在 ANKRD24 中发现了一个新的同源框移变异 c.1934_1937del; (p.Thr645Lysfs*52),该变异与该家族的 HL 表型存在分离关系。最近的研究表明,ANKRD24 是耳蜗毛细胞立体纤毛根小的关键成分,它与 TRIOBP 相互作用,而 TRIOBP 蛋白已经与人类耳聋有牵连。我们的数据首次表明 ANKRD24 与人类非综合征性 HL(NSHL)有关,并扩大了 HL 的遗传范围。
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A Frameshift Variant in ANKRD24 Implicates Its Role in Human Non-Syndromic Hearing Loss.

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.

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来源期刊
Clinical Genetics
Clinical Genetics 医学-遗传学
CiteScore
6.50
自引率
0.00%
发文量
175
审稿时长
3-8 weeks
期刊介绍: Clinical Genetics links research to the clinic, translating advances in our understanding of the molecular basis of genetic disease for the practising clinical geneticist. The journal publishes high quality research papers, short reports, reviews and mini-reviews that connect medical genetics research with clinical practice. Topics of particular interest are: • Linking genetic variations to disease • Genome rearrangements and disease • Epigenetics and disease • The translation of genotype to phenotype • Genetics of complex disease • Management/intervention of genetic diseases • Novel therapies for genetic diseases • Developmental biology, as it relates to clinical genetics • Social science research on the psychological and behavioural aspects of living with or being at risk of genetic disease
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