Literature DB >> 25500575

Lethal neonatal rigidity and multifocal seizure syndrome--report of another family with a BRAT1 mutation.

Rachel Straussberg1, Esther Ganelin-Cohen2, Hadassah Goldberg-Stern3, Shay Tzur4, Doron M Behar5, Pola Smirin-Yosef6, Mali Salmon-Divon7, Lina Basel-Vanagaite8.   

Abstract

We describe two siblings born to consanguineous Arab-Muslim parents who presented in early infancy with myoclonic seizures, hypertonia and contractures, arrested head growth, inability to swallow, and bouts of apnea-bradycardia, culminating in cardiac arrest and death. Whole-genome sequencing yielded a c.1173delG mutation in the BRAT1 gene. Three recent reports identified mutations in the same gene in three infants from three Amish sibships, one Mexican neonate and two Japanese siblings with similar clinical manifestations. The authors speculated that the destabilization of the encoded protein may underlie the catastrophic epilepsy and corticobasal neuronal degeneration. We suggest that BRAT1 be added to the growing list of genes that are related to severe early infantile (neonatal) epileptic encephalopathy.
Copyright © 2014 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Apnea; Bradycardia; Epilepsy; Hypertonicity; Infancy; Seizures

Mesh:

Substances:

Year:  2014        PMID: 25500575     DOI: 10.1016/j.ejpn.2014.11.004

Source DB:  PubMed          Journal:  Eur J Paediatr Neurol        ISSN: 1090-3798            Impact factor:   3.140


  8 in total

1.  A review of the clinical spectrum of BRAT1 disorders and case of developmental and epileptic encephalopathy surviving into adulthood.

Authors:  Ross Fowkes; Menatalla Elwan; Ela Akay; Clinton J Mitchell; Rhys H Thomas; David Lewis-Smith
Journal:  Epilepsy Behav Rep       Date:  2022-05-08

2.  Mutations in BRAT1 cause autosomal recessive progressive encephalopathy: Report of a Spanish patient.

Authors:  Alberto Fernández-Jaén; Sara Álvarez; Eui Young So; Toru Ouchi; Mar Jiménez de la Peña; Anna Duat; Daniel Martín Fernández-Mayoralas; Ana Laura Fernández-Perrone; Jacobo Albert; Beatriz Calleja-Pérez
Journal:  Eur J Paediatr Neurol       Date:  2016-02-21       Impact factor: 3.140

Review 3.  BRAT1 mutations present with a spectrum of clinical severity.

Authors:  Siddharth Srivastava; Heather E Olson; Julie S Cohen; Cynthia S Gubbels; Sharyn Lincoln; Brigette Tippin Davis; Layla Shahmirzadi; Siddharth Gupta; Jonathan Picker; Timothy W Yu; David T Miller; Janet S Soul; Andrea Poretti; SakkuBai Naidu
Journal:  Am J Med Genet A       Date:  2016-06-09       Impact factor: 2.802

4.  Clinical care models in the era of next-generation sequencing.

Authors:  Anne Slavotinek
Journal:  Mol Genet Genomic Med       Date:  2016-05-12       Impact factor: 2.183

5.  Lethal neonatal rigidity and multifocal seizure syndrome with a new mutation in BRAT1.

Authors:  Yalcin Celik; Cetin Okuyaz; Ali Ertug Arslankoylu; Serdar Ceylaner
Journal:  Epilepsy Behav Case Rep       Date:  2017-05-25

6.  Novel Biallelic Variant in the BRAT1 Gene Caused Nonprogressive Cerebellar Ataxia Syndrome.

Authors:  Yiming Qi; Xueqi Ji; Hongke Ding; Ling Liu; Yan Zhang; Aihua Yin
Journal:  Front Genet       Date:  2022-03-10       Impact factor: 4.599

7.  BRAT1 links Integrator and defective RNA processing with neurodegeneration.

Authors:  Zuzana Cihlarova; Jan Kubovciak; Margarita Sobol; Katerina Krejcikova; Jana Sachova; Michal Kolar; David Stanek; Cyril Barinka; Grace Yoon; Keith W Caldecott; Hana Hanzlikova
Journal:  Nat Commun       Date:  2022-08-26       Impact factor: 17.694

8.  An intronic variant in BRAT1 creates a cryptic splice site, causing epileptic encephalopathy without prominent rigidity.

Authors:  Fatma Kurt Colak; Naz Guleray; Ebru Azapagasi; Mutlu Uysal Yazıcı; Erhan Aksoy; Nesrin Ceylan
Journal:  Acta Neurol Belg       Date:  2020-10-10       Impact factor: 2.396

  8 in total

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