Literature DB >> 29801887

A study of Huntington disease-like syndromes in black South African patients reveals a single SCA2 mutation and a unique distribution of normal alleles across five repeat loci.

Fiona K Baine1, Nabeelah Peerbhai2, Amanda Krause2.   

Abstract

Huntington disease (HD) is a progressive neurodegenerative disease, characterised by a triad of movement disorder, emotional and behavioural disturbances and cognitive impairment. The underlying cause is an expanded CAG repeat in the huntingtin gene. For a small proportion of patients presenting with HD-like symptoms, the mutation in this gene is not identified and they are said to have a HD "phenocopy". South Africa has the highest number of recorded cases of an African-specific phenocopy, Huntington disease-like 2 (HDL2), caused by a repeat expansion in the junctophilin-3 gene. However, a significant proportion of black patients with clinical symptoms suggestive of HD still test negative for HD and HDL2. This study thus aimed to investigate five other loci associated with HD phenocopy syndromes - ATN1, ATXN2, ATXN7, TBP and C9orf72. In a sample of patients in whom HD and HDL2 had been excluded, a single expansion was identified in the ATXN2 gene, confirming a diagnosis of Spinocerebellar ataxia 2. The results indicate that common repeat expansion disorders do not contribute significantly to the HD-like phenotype in black South African patients. Importantly, allele sizing reveals unique distributions of normal repeat lengths across the associated loci in the African population studied.
Copyright © 2018 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Africa; HD; HD phenocopies; HDL2; Huntington disease; Huntington disease-like 2; Phenocopy; Polyglutamine; Repeat expansions; South Africa

Mesh:

Substances:

Year:  2018        PMID: 29801887     DOI: 10.1016/j.jns.2018.04.031

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  5 in total

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Journal:  J Neurol Sci       Date:  2019-04-17       Impact factor: 3.181

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4.  Repeats expansions in ATXN2, NOP56, NIPA1 and ATXN1 are not associated with ALS in Africans.

Authors:  Melissa Nel; Thandeka Mavundla; Kayleigh Gultig; Gerrit Botha; Nicola Mulder; Michael Benatar; Joanne Wuu; Anne Cooley; Jason Myers; Evadnie Rampersaud; Gang Wu; Jeannine M Heckmann
Journal:  IBRO Neurosci Rep       Date:  2021-02-10

5.  C9orf72 Hexanucleotide Repeat in Huntington-Like Patients: Systematic Review and Meta-Analysis.

Authors:  Carlos Alva-Diaz; Christoper A Alarcon-Ruiz; Kevin Pacheco-Barrios; Nicanor Mori; Josmel Pacheco-Mendoza; Bryan J Traynor; Andrea Rivera-Valdivia; Pongtawat Lertwilaiwittaya; Thomas D Bird; Mario Cornejo-Olivas
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  5 in total

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