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与摩洛哥家族非综合征性耳聋相关的FOXI1和TMPRSS3基因纯合错义变体

Homozygous Missense Variants in FOXI1 and TMPRSS3 Genes Associated with Non-syndromic Deafness in Moroccan Families.

作者信息

AitRaise Imane, Amalou Ghita, Bakhchane Amina, Bousfiha Amale, Abdelghaffar Houria, Majida Charif, Bonnet Crystel, Petit Christine, Barakat Abdelhamid

机构信息

Genomics and Human Genetics Laboratory, Institut Pasteur du Maroc, Casablanca, Morocco.

Laboratory of Biochemistry, Environment and Agri-food, Faculty of Science and Techniques of Mohammedia, Hassan II University of Casablanca, Casablanca, Morocco.

出版信息

Biochem Genet. 2024 Jun;62(3):1914-1924. doi: 10.1007/s10528-023-10515-5. Epub 2023 Oct 1.

DOI:10.1007/s10528-023-10515-5
PMID:37777971
Abstract

One of the most prevalent sensorineural disorders, autosomal recessive non-syndromic hearing loss (ARNSHL) which can affect all age groups, from the newborn (congenital) to the elderly (presbycusis). Important etiologic, phenotypic, and genotypic factors can cause deafness. So far, the high genetic variability that explains deafness makes molecular diagnosis challenging. In Morocco, the GJB2 gene is the primary cause of non-syndromic hereditary deafness, while the existence of a variant in the LRTOMT gene is the second cause of this condition. After excluding these two frequently occurring GJB2 and LRTOMT variants, whole-exome sequencing was carried out in two Moroccan consanguineous families with hearing loss. As a result, two novel variants in the TMPRSS3 (c.1078G>A, p. Ala 360Thr) and FOXI1 (c.6C>G, p. Ser 2Arg) genes have been discovered in deaf patients and the pathogenic effect has been anticipated by several bioinformatics and molecular modeling systems. For the first time, these variants are identified in the Moroccan population, showing the population heterogeneity and demonstrating the value of the WES in hearing loss diagnosis.

摘要

常染色体隐性非综合征性听力损失(ARNSHL)是最常见的感音神经性疾病之一,可影响从新生儿(先天性)到老年人(老年性耳聋)的所有年龄组。重要的病因、表型和基因型因素均可导致耳聋。到目前为止,导致耳聋的高遗传变异性使得分子诊断具有挑战性。在摩洛哥,GJB2基因是非综合征性遗传性耳聋的主要原因,而LRTOMT基因变异的存在是导致这种情况的第二个原因。在排除这两种常见的GJB2和LRTOMT变异后,对两个患有听力损失的摩洛哥近亲家庭进行了全外显子组测序。结果,在耳聋患者中发现了TMPRSS3(c.1078G>A,p.Ala 360Thr)和FOXI1(c.6C>G,p.Ser 2Arg)基因中的两个新变异,并且几个生物信息学和分子建模系统已经预测了其致病作用。这些变异首次在摩洛哥人群中被鉴定出来,显示了人群异质性,并证明了全外显子组测序在听力损失诊断中的价值。

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本文引用的文献

1
Structural analysis of pathogenic TMPRSS3 variants and their cochlear implantation outcomes of sensorineural hearing loss.致病性TMPRSS3变体的结构分析及其感音神经性听力损失的人工耳蜗植入结果
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Analysis of SLC26A4, FOXI1, and KCNJ10 Gene Variants in Patients with Incomplete Partition of the Cochlea and Enlarged Vestibular Aqueduct (EVA) Anomalies.
分析不完全分隔耳蜗和扩大前庭水管(EVA)异常患者的 SLC26A4、FOXI1 和 KCNJ10 基因突变。
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