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全外显子组测序揭示与肾病综合征相关的 COQ8B 基因的新型纯合突变。

Whole-exome sequencing reveals a novel homozygous mutation in the COQ8B gene associated with nephrotic syndrome.

机构信息

PK-PD Formulation and Toxicology Division, CSIR Indian Institute of Integrative Medicine, Canal Road, Jammu, 180001, India.

Academy of Scientific & Innovative Research (AcSIR), Ghaziabad, Uttar Pradesh, 201002, India.

出版信息

Sci Rep. 2021 Jun 25;11(1):13337. doi: 10.1038/s41598-021-92023-3.

Abstract

Nephrotic syndrome arising from monogenic mutations differs substantially from acquired ones in their clinical prognosis, progression, and disease management. Several pathogenic mutations in the COQ8B gene are known to cause nephrotic syndrome. Here, we used the whole-exome sequencing (WES) technology to decipher the genetic cause of nephrotic syndrome (CKD stage-V) in a large affected consanguineous family. Our study exposed a novel missense homozygous mutation NC_000019.9:g.41209497C > T; NM_024876.4:c.748G > A; NP_079152.3:p.(Asp250Asn) in the 9th exon of the COQ8B gene, co-segregated well with the disease phenotype. Our study provides the first insight into this homozygous condition, which has not been previously reported in 1000Genome, ClinVar, ExAC, and genomAD databases. In addition to the pathogenic COQ8B variant, the WES data also revealed some novel and recurrent mutations in the GLA, NUP107, COQ2, COQ6, COQ7 and COQ9 genes. The novel variants observed in this study have been submitted to the ClinVar database and are publicly available online with the accessions: SCV001451361.1, SCV001451725.1 and SCV001451724.1. Based on the patient's clinical history and genomic data with in silico validation, we conclude that pathogenic mutation in the COQ8B gene was causing kidney failure in an autosomal recessive manner. We recommend WES technology for genetic testing in such a consanguineous family to not only prevent the future generation, but early detection can help in disease management and therapeutic interventions.

摘要

由单基因突变引起的肾病综合征在其临床预后、进展和疾病管理方面与获得性肾病综合征有很大的不同。已知 COQ8B 基因中的几个致病突变可导致肾病综合征。在这里,我们使用全外显子组测序 (WES) 技术来破译一个大型近亲家族中肾病综合征(CKD 期-V)的遗传原因。我们的研究揭示了 COQ8B 基因第 9 外显子中的一个新的错义纯合突变 NC_000019.9:g.41209497C>T; NM_024876.4:c.748G>A; NP_079152.3:p.(Asp250Asn),与疾病表型很好地共分离。我们的研究首次深入了解了这种纯合状态,该状态在 1000Genome、ClinVar、ExAC 和 genomAD 数据库中均未报道过。除了致病的 COQ8B 变体外,WES 数据还揭示了 GLA、NUP107、COQ2、COQ6、COQ7 和 COQ9 基因中的一些新的和反复出现的突变。本研究中观察到的新变体已提交 ClinVar 数据库,可在线公开获取,访问号为:SCV001451361.1、SCV001451725.1 和 SCV001451724.1。基于患者的临床病史和基因组数据以及计算机模拟验证,我们得出结论,COQ8B 基因中的致病性突变以常染色体隐性方式导致肾功能衰竭。我们建议在这种近亲家庭中使用 WES 技术进行基因检测,不仅可以预防下一代,而且早期检测还可以帮助进行疾病管理和治疗干预。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5f0e/8233304/f42764cd504c/41598_2021_92023_Fig1_HTML.jpg

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