Literature DB >> 31857255

A novel homozygous splice-site mutation in the SPTBN4 gene causes axonal neuropathy without intellectual disability.

Martin G Häusler1, Matthias Begemann2, Hart G Lidov3, Ingo Kurth2, Basil T Darras4, Miriam Elbracht2.   

Abstract

Mutations in spectrin beta non-erythrocytic 4 (SPTBN4) have been linked to congenital hypotonia, intellectual disability and motor neuropathy. Here we report on two siblings with a homozygous splice-site mutation in the SPTBN4 gene, lacking previously reported features of the disorder such as seizures, feeding difficulties, respiratory difficulties or profound intellectual disability. Our findings indicate that muscular hypotonia, myopathic facies with ptosis and axonal neuropathy can be the core clinical features in the SPTBN4 disorder and suggest that SPTBN4 mutation analysis should be considered in infants with marked axonal neuropathy.
Copyright © 2019 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Axonal neuropathy; Ptosis; SPTBN4; Spectrin beta non-erythrocytic 4

Year:  2019        PMID: 31857255     DOI: 10.1016/j.ejmg.2019.103826

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  4 in total

Review 1.  Regulation of Cardiac Conduction and Arrhythmias by Ankyrin/Spectrin-Based Macromolecular Complexes.

Authors:  Drew Nassal; Jane Yu; Dennison Min; Cemantha Lane; Rebecca Shaheen; Daniel Gratz; Thomas J Hund
Journal:  J Cardiovasc Dev Dis       Date:  2021-04-29

2.  A genome-wide association study in human lymphoblastoid cells supports safety of mitochondrial complex I inhibitor.

Authors:  Huanyao Gao; Utkarsh Tripathi; Sergey Trushin; Lela Okromelidze; Nicholas P Pichurin; Lixuan Wei; Yongxian Zhuang; Liewei Wang; Eugenia Trushina
Journal:  Mitochondrion       Date:  2021-02-19       Impact factor: 4.160

3.  Novel bi-allelic variants expand the SPTBN4-related genetic and phenotypic spectrum.

Authors:  Markus Buelow; David Süßmuth; Laurie D Smith; Omid Aryani; Claudia Castiglioni; Werner Stenzel; Enrico Bertini; Markus Schuelke; Ellen Knierim
Journal:  Eur J Hum Genet       Date:  2021-03-26       Impact factor: 4.246

4.  Pathogenic SPTBN1 variants cause an autosomal dominant neurodevelopmental syndrome.

Authors:  Margot A Cousin; Blake A Creighton; Keith A Breau; Rebecca C Spillmann; Erin Torti; Sruthi Dontu; Swarnendu Tripathi; Deepa Ajit; Reginald J Edwards; Simone Afriyie; Julia C Bay; Kathryn M Harper; Alvaro A Beltran; Lorena J Munoz; Liset Falcon Rodriguez; Michael C Stankewich; Richard E Person; Yue Si; Elizabeth A Normand; Amy Blevins; Alison S May; Louise Bier; Vimla Aggarwal; Grazia M S Mancini; Marjon A van Slegtenhorst; Kirsten Cremer; Jessica Becker; Hartmut Engels; Stefan Aretz; Jennifer J MacKenzie; Eva Brilstra; Koen L I van Gassen; Richard H van Jaarsveld; Renske Oegema; Gretchen M Parsons; Paul Mark; Ingo Helbig; Sarah E McKeown; Robert Stratton; Benjamin Cogne; Bertrand Isidor; Pilar Cacheiro; Damian Smedley; Helen V Firth; Tatjana Bierhals; Katja Kloth; Deike Weiss; Cecilia Fairley; Joseph T Shieh; Amy Kritzer; Parul Jayakar; Evangeline Kurtz-Nelson; Raphael A Bernier; Tianyun Wang; Evan E Eichler; Ingrid M B H van de Laar; Allyn McConkie-Rosell; Marie T McDonald; Jennifer Kemppainen; Brendan C Lanpher; Laura E Schultz-Rogers; Lauren B Gunderson; Pavel N Pichurin; Grace Yoon; Michael Zech; Robert Jech; Juliane Winkelmann; Adriana S Beltran; Michael T Zimmermann; Brenda Temple; Sheryl S Moy; Eric W Klee; Queenie K-G Tan; Damaris N Lorenzo
Journal:  Nat Genet       Date:  2021-07-01       Impact factor: 41.307

  4 in total

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