Literature DB >> 36190665

A novel biallelic variant further delineates PRDX3-related autosomal recessive cerebellar ataxia.

Misbahuddin M Rafeeq1, Muhammad Umair2,3, Muhammad Bilal4, Alaa Hamed Habib1, Ahmed Waqas5, Ziaullah M Sain6, Mohammad Zubair Alam7,8, Raja Hussain Ali9.   

Abstract

Cerebellar ataxias (CAs) comprise a rare group of neurological disorders characterized by extensive phenotypic and genetic heterogeneity. In the last several years, our understanding of the CA etiology has increased significantly and resulted in the discoveries of numerous ataxia-associated genes. Herein, we describe a single affected individual from a consanguineous family segregating a recessive neurodevelopmental disorder. The proband showed features such as global developmental delay, cerebellar atrophy, hypotonia, speech issues, dystonia, and profound hearing impairment. Whole-exome sequencing and Sanger sequencing revealed a biallelic nonsense variant (c.496A > T; p.Lys166*) in the exon 5 of the PRDX3 gene that segregated perfectly within the family. This is the third report that associates the PRDX3 gene variant with cerebellar ataxia. In addition, associated hearing impairment further delineates the PRDX3 associated gene phenotypes.
© 2022. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Entities:  

Keywords:  Cerebellar ataxia; Nonsense variant; Novel variant; PRDX3; Profound deafness; Whole-exome sequencing

Year:  2022        PMID: 36190665     DOI: 10.1007/s10048-022-00701-9

Source DB:  PubMed          Journal:  Neurogenetics        ISSN: 1364-6745            Impact factor:   3.017


  6 in total

1.  Biallelic loss-of-function variations in PRDX3 cause cerebellar ataxia.

Authors:  Adriana P Rebelo; Ilse Eidhof; Vivian P Cintra; Léna Guillot-Noel; Claudia V Pereira; Dagmar Timmann; Andreas Traschütz; Ludger Schöls; Giulia Coarelli; Alexandra Durr; Mathieu Anheim; Christine Tranchant; Bart van de Warrenburg; Claire Guissart; Michel Koenig; Jack Howell; Carlos T Moraes; Annette Schenck; Giovanni Stevanin; Stephan Züchner; Matthis Synofzik
Journal:  Brain       Date:  2021-04-23       Impact factor: 13.501

2.  First direct evidence of involvement of a homozygous loss-of-function variant in the EPS15L1 gene underlying split-hand/split-foot malformation.

Authors:  M Umair; A Ullah; S Abbas; F Ahmad; S Basit; W Ahmad
Journal:  Clin Genet       Date:  2018-01-25       Impact factor: 4.438

3.  Biallelic variant in DACH1, encoding Dachshund Homolog 1, defines a novel candidate locus for recessive postaxial polydactyly type A.

Authors:  Muhammad Umair; Oliva Palander; Muhammad Bilal; Bader Almuzzaini; Qamre Alam; Farooq Ahmad; Muhammad Younus; Amjad Khan; Ahmed Waqas; Misbahuddin M Rafeeq; Majid Alfadhel
Journal:  Genomics       Date:  2021-05-20       Impact factor: 5.736

Review 4.  Hereditary ataxias: overview.

Authors:  Suman Jayadev; Thomas D Bird
Journal:  Genet Med       Date:  2013-03-28       Impact factor: 8.822

Review 5.  Peroxiredoxins-The Underrated Actors during Virus-Induced Oxidative Stress.

Authors:  Inna L Karpenko; Vladimir T Valuev-Elliston; Olga N Ivanova; Olga A Smirnova; Alexander V Ivanov
Journal:  Antioxidants (Basel)       Date:  2021-06-18

Review 6.  Peroxiredoxins and the Regulation of Cell Death.

Authors:  Mark B Hampton; Karina M O'Connor
Journal:  Mol Cells       Date:  2016-01-25       Impact factor: 5.034

  6 in total

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