Isoform-specific loss of dystonin causes hereditary motor and sensory neuropathy.


Journal

Neurology. Genetics
ISSN: 2376-7839
Titre abrégé: Neurol Genet
Pays: United States
ID NLM: 101671068

Informations de publication

Date de publication:
Oct 2020
Historique:
received: 08 03 2020
accepted: 28 05 2020
entrez: 18 8 2020
pubmed: 18 8 2020
medline: 18 8 2020
Statut: epublish

Résumé

To determine the genetic cause of axonal Charcot-Marie-Tooth disease in a small family with 2 affected siblings, one of whom had cerebellar features on examination. Whole-exome sequencing of genomic DNA and analysis for recessively inherited mutations; PCR-based messenger RNA/complementary DNA analysis of transcripts to characterize the effects of variants identified by exome sequencing. We identified compound heterozygous mutations in dystonin ( These findings introduce a novel human phenotype, axonal Charcot-Marie-Tooth, of recessive

Identifiants

pubmed: 32802955
doi: 10.1212/NXG.0000000000000496
pii: NG2020013508
pmc: PMC7413632
doi:

Types de publication

Journal Article

Langues

eng

Pagination

e496

Informations de copyright

Copyright © 2020 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology.

Références

Neurol Genet. 2020 Jan 02;6(1):e389
pubmed: 32042917
Dev Genet. 1998;22(2):160-8
pubmed: 9581287
Nature. 1988 Sep 29;335(6189):435-7
pubmed: 3138544
Hum Mutat. 2015 Oct;36(10):950-6
pubmed: 26173844
Proc Natl Acad Sci U S A. 2007 Feb 13;104(7):2223-8
pubmed: 17287360
Hum Mol Genet. 2014 May 15;23(10):2694-710
pubmed: 24381311
Mol Biol Cell. 2012 Feb;23(4):553-66
pubmed: 22190742
Hum Mol Genet. 2015 Mar 1;24(5):1267-79
pubmed: 25348330
PLoS One. 2011;6(6):e21093
pubmed: 21698255
Neurology. 2017 May 30;88(22):2132-2140
pubmed: 28468842
Nat Rev Neurol. 2013 Oct;9(10):562-71
pubmed: 24018473
J Invest Dermatol. 2012 Mar;132(3 Pt 1):742-4
pubmed: 22113475
Br J Dermatol. 2015 Feb;172(2):527-31
pubmed: 25059916
Am J Med Genet A. 2020 Jan;182(1):268
pubmed: 31633269
J Mol Biol. 2007 Feb 9;366(1):244-57
pubmed: 17161423
Exp Cell Res. 2007 Jun 10;313(10):2189-203
pubmed: 17499243
Neuroscience. 1988 Nov;27(2):403-24
pubmed: 3217000
Bioinformatics. 2010 Mar 1;26(5):589-95
pubmed: 20080505
Ann Neurol. 2012 Apr;71(4):569-72
pubmed: 22522446
Genome Res. 2010 Sep;20(9):1297-303
pubmed: 20644199
Hum Mutat. 2019 Jan;40(1):106-114
pubmed: 30371979
J Peripher Nerv Syst. 2011 Sep;16(3):191-8
pubmed: 22003934
Nat Genet. 1995 Jul;10(3):301-6
pubmed: 7670468
Curr Opin Cell Biol. 2004 Jun;16(3):293-9
pubmed: 15145354
J Clin Invest. 1988 Dec;82(6):1864-70
pubmed: 2461961
Am J Med Genet A. 2017 Oct;173(10):2743-2746
pubmed: 28767192
Exp Cell Res. 2006 Sep 10;312(15):2712-25
pubmed: 16797530
Brain. 1964 Jun;87:367-78
pubmed: 14188280
J Invest Dermatol. 2010 Jun;130(6):1551-7
pubmed: 20164846
J Cell Biol. 2003 Oct 27;163(2):223-9
pubmed: 14581450
Ann Neurol. 2011 Jan;69(1):22-33
pubmed: 21280073
Cell Motil Cytoskeleton. 2007 Dec;64(12):897-905
pubmed: 17849487

Auteurs

William W Motley (WW)

Department of Neurology (W.W.M., S.S.S.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; and Department of Human Genetics (S.Z.), Hussman Institute for Human Genomics, University of Miami, FL.

Stephan Züchner (S)

Department of Neurology (W.W.M., S.S.S.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; and Department of Human Genetics (S.Z.), Hussman Institute for Human Genomics, University of Miami, FL.

Steven S Scherer (SS)

Department of Neurology (W.W.M., S.S.S.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; and Department of Human Genetics (S.Z.), Hussman Institute for Human Genomics, University of Miami, FL.

Classifications MeSH