Literature DB >> 32948840

Expansion of the GRIA2 phenotypic representation: a novel de novo loss of function mutation in a case with childhood onset schizophrenia.

Anna Alkelai1, Shahar Shohat2, Lior Greenbaum3,4,5, Tanya Schechter6, Benjamin Draiman6, Eti Chitrit-Raveh6, Shlomit Rienstein3, Neha Dagaonkar7, Daniel Hughes7, Vimla S Aggarwal7, Erin L Heinzen7,8, Sagiv Shifman2, David B Goldstein7, Yoav Kohn6,9.   

Abstract

Childhood-onset schizophrenia (COS) is a rare form of schizophrenia with an onset before 13 years of age. There is rising evidence that genetic factors play a major role in COS etiology, yet, only a few single gene mutations have been discovered. Here we present a diagnostic whole-exome sequencing (WES) in an Israeli Jewish female with COS and additional neuropsychiatric conditions such as obsessive-compulsive disorder (OCD), anxiety, and aggressive behavior. Variant analysis revealed a de novo novel stop gained variant in GRIA2 gene (NM_000826.4: c.1522 G > T (p.Glu508Ter)). GRIA2 encodes for a subunit of the AMPA sensitive glutamate receptor (GluA2) that functions as ligand-gated ion channel in the central nervous system and plays an important role in excitatory synaptic transmission. GluA2 subunit mutations are known to cause variable neurodevelopmental phenotypes including intellectual disability, autism spectrum disorder, epilepsy, and OCD. Our findings support the potential diagnostic role of WES in COS, identify GRIA2 as possible cause to a broad psychiatric phenotype that includes COS as a major manifestation and expand the previously reported GRIA2 loss of function phenotypes.

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Year:  2020        PMID: 32948840     DOI: 10.1038/s10038-020-00846-1

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  1 in total

1.  Exploration and characterisation of the phenotypic and genetic profiles of patients with early onset schizophrenia associated with autism spectrum disorder and their first-degree relatives: a French multicentre case series study protocol (GenAuDiss).

Authors:  Arnaud Fernandez; Emmanuelle Dor; Thomas Maurin; Gaelle Laure; Marie-Line Menard; Małgorzata Drozd; Francois Poinso; Barbara Bardoni; Florence Askenazy; Susanne Thümmler
Journal:  BMJ Open       Date:  2018-07-05       Impact factor: 2.692

  1 in total
  3 in total

1.  The benefit of diagnostic whole genome sequencing in schizophrenia and other psychotic disorders.

Authors:  Anna Alkelai; Lior Greenbaum; Anna R Docherty; Andrey A Shabalin; Gundula Povysil; Ayan Malakar; Daniel Hughes; Shannon L Delaney; Emma P Peabody; James McNamara; Sahar Gelfman; Evan H Baugh; Anthony W Zoghbi; Matthew B Harms; Hann-Shyan Hwang; Anat Grossman-Jonish; Vimla Aggarwal; Erin L Heinzen; Vaidehi Jobanputra; Ann E Pulver; Bernard Lerer; David B Goldstein
Journal:  Mol Psychiatry       Date:  2021-11-19       Impact factor: 13.437

2.  Case Report: A Novel De Novo Missense Mutation of the GRIA2 Gene in a Chinese Case of Neurodevelopmental Disorder With Language Impairment.

Authors:  Bingbo Zhou; Chuan Zhang; Lei Zheng; Zhiqiang Wang; Xue Chen; Xuan Feng; Qinghua Zhang; Shengju Hao; Liwan Wei; Weiyue Gu; Ling Hui
Journal:  Front Genet       Date:  2021-11-25       Impact factor: 4.599

3.  Identification and functional evaluation of GRIA1 missense and truncation variants in individuals with ID: An emerging neurodevelopmental syndrome.

Authors:  Vardha Ismail; Linda G Zachariassen; Annie Godwin; Mane Sahakian; Sian Ellard; Karen L Stals; Emma Baple; Kate Tatton Brown; Nicola Foulds; Gabrielle Wheway; Matthew O Parker; Signe M Lyngby; Miriam G Pedersen; Julie Desir; Allan Bayat; Maria Musgaard; Matthew Guille; Anders S Kristensen; Diana Baralle
Journal:  Am J Hum Genet       Date:  2022-06-07       Impact factor: 11.043

  3 in total

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