Literature DB >> 34172776

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

Mohd Fareed1,2, Vikas Makkar3, Ravi Angral4, Mohammad Afzal5, Gurdarshan Singh6,7.   

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.

Entities:  

Year:  2021        PMID: 34172776     DOI: 10.1038/s41598-021-92023-3

Source DB:  PubMed          Journal:  Sci Rep        ISSN: 2045-2322            Impact factor:   4.379


  1 in total

1.  The neXtProt knowledgebase in 2020: data, tools and usability improvements.

Authors:  Monique Zahn-Zabal; Pierre-André Michel; Alain Gateau; Frédéric Nikitin; Mathieu Schaeffer; Estelle Audot; Pascale Gaudet; Paula D Duek; Daniel Teixeira; Valentine Rech de Laval; Kasun Samarasinghe; Amos Bairoch; Lydie Lane
Journal:  Nucleic Acids Res       Date:  2020-01-08       Impact factor: 16.971

  1 in total
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1.  COQ8B glomerular nephropathy: Outcomes after kidney transplantation and analysis of characteristics in Chinese population.

Authors:  Shuhan Zeng; Yuanyuan Xu; Cheng Cheng; Nannan Yu; Longshan Liu; Ying Mo; Lizhi Chen; Xiaoyun Jiang
Journal:  Front Pediatr       Date:  2022-08-10       Impact factor: 3.569

  1 in total

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