一个常染色体显性遗传性听力损失家族中发现的TECTA c.6183G>T变体的临床相关性:一例报告。

IF 1.5 4区 医学 Q2 MEDICINE, GENERAL & INTERNAL Croatian Medical Journal Pub Date : 2023-10-31
Ivona Sansović, Ana-Maria Meašić, Ljubica Odak, Mijana Kero
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引用次数: 0

摘要

α-检测素基因(TECTA)的错义变异导致常染色体显性遗传(DFNA8/A12)非综合征性听力损失(ADNSHL),并导致相当多的ADNSHL病例。根据基因型-表型相关性研究,α-检测器蛋白透明带(ZP)结构域的错义变体主要导致中频HL。在这里,我们报道了一个患有早发性、感觉神经性、中度至重度中频HL的大家族的临床外显子组测序结果。我们在TECTA的ZP结构域附近发现了一个杂合的c.6183G>T变体,在五个家族成员中分离。该变体先前被报道为ADNSHL家族中具有不确定意义的变体。根据目前研究的家族中的特定分离和美国医学遗传学与基因组学学院的一般指南,我们认为TECTA c.6183G>T变体应被视为ADNSHL的可能致病原因。本报告增加了对罕见的c.6183G>T错义变体的了解,该变体影响TECTA中ZP结构域的紧邻区域。我们的研究结果强调了家族性HL患者的临床评估以及在评估变体致病性时研究家族分离的重要性。
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Clinical relevance of the TECTA c.6183G>T variant identified in a family with autosomal dominant hearing loss: a case report.

Missense variants in the α-tectorin gene (TECTA) cause autosomal dominant (DFNA8/A12) non-syndromic hearing loss (ADNSHL) and account for a considerable number of ADNSHL cases. According to genotype-phenotype correlation studies, missense variants in the zona pellucida (ZP) domain of α-tectorin predominantly cause mid-frequency HL. Here, we report on clinical exome sequencing results in a large family with early-onset, sensorineural, moderate-to-severe mid-frequency HL. We identified one heterozygous c.6183G>T variant near the ZP domain of TECTA segregating in five family members. This variant was previously reported as a variant of uncertain significance in a family with ADNSHL. On the basis of specific segregation in the currently studied family and the general guidelines of the American College of Medical Genetics and Genomics, we argue that the TECTA c.6183G>T variant should be considered a likely pathogenic cause of ADNSHL. This report adds to the knowledge on the rare c.6183G>T missense variant, which affects the immediate vicinity of the ZP domain in TECTA. Our findings highlight the importance of clinical evaluation in patients with familial HL and of studying family segregation when assessing the pathogenicity of a variant.

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来源期刊
Croatian Medical Journal
Croatian Medical Journal 医学-医学:内科
CiteScore
3.00
自引率
5.30%
发文量
105
审稿时长
6-12 weeks
期刊介绍: Croatian Medical Journal (CMJ) is an international peer reviewed journal open to scientists from all fields of biomedicine and health related research. Although CMJ welcomes all contributions that increase and expand on medical knowledge, the two areas are of the special interest: topics globally relevant for biomedicine and health and medicine in developing and emerging countries.
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