% IMPORTANT: The following is UTF-8 encoded. This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.
@ARTICLE{Pellerin:276089,
author = {Pellerin, David and Iruzubieta, Pablo and Xu, Isaac R L and
Danzi, Matt C and Cortese, Andrea and Synofzik, Matthis and
Houlden, Henry and Zuchner, Stephan and Brais, Bernard},
title = {{R}ecent {A}dvances in the {G}enetics of {A}taxias: {A}n
{U}pdate on {N}ovel {A}utosomal {D}ominant {R}epeat
{E}xpansions.},
journal = {Current neurology and neuroscience reports},
volume = {25},
number = {1},
issn = {1528-4042},
address = {Philadelphia, Pa.},
publisher = {Current Science Inc.},
reportid = {DZNE-2025-00170},
pages = {16},
year = {2025},
abstract = {Autosomal dominant cerebellar ataxias, also known as
spinocerebellar ataxias (SCAs), are genetically and
clinically diverse neurodegenerative disorders characterized
by progressive cerebellar dysfunction. Despite advances in
sequencing technologies, a large proportion of patients with
SCA still lack a definitive genetic diagnosis. The advent of
advanced bioinformatic tools and emerging genomics
technologies, such as long-read sequencing, offers an
unparalleled opportunity to close the diagnostic gap for
hereditary ataxias. This article reviews the recently
identified repeat expansion SCAs and describes their
molecular basis, epidemiology, and clinical
features.Leveraging advanced bioinformatic tools and
long-read sequencing, recent studies have identified novel
pathogenic short tandem repeat expansions in FGF14, ZFHX3,
and THAP11, associated with SCA27B, SCA4, and SCA51,
respectively. SCA27B, caused by an intronic (GAA)•(TTC)
repeat expansion, has emerged as one of the most common
forms of adult-onset hereditary ataxias, especially in
European populations. The coding GGC repeat expansion in
ZFHX3 causing SCA4 was identified more than 25 years after
the disorder's initial clinical description and appears to
be a rare cause of ataxia outside northern Europe. SCA51,
caused by a coding CAG repeat expansion, is overall rare and
has been described in a small number of patients. The recent
identification of three novel pathogenic repeat expansions
underscores the importance of this class of genomic
variation in the pathogenesis of SCAs. Progress in
sequencing technologies holds promise for closing the
diagnostic gap in SCAs and guiding the development of
therapeutic strategies for ataxia.},
subtyp = {Review Article},
keywords = {Humans / Spinocerebellar Ataxias: genetics /
Spinocerebellar Ataxias: diagnosis / DNA Repeat Expansion:
genetics / Autosomal dominant cerebellar ataxia (Other) /
FGF14 (Other) / Genome sequencing (Other) / Spinocerebellar
ataxia (Other) / Spinocerebellar ataxia 27B (SCA27B) (Other)
/ Spinocerebellar ataxia 4 (SCA4) (Other) / Spinocerebellar
ataxia 51 (SCA51) (Other) / THAP11 (Other) / ZFHX3 (Other)},
cin = {AG Gasser},
ddc = {610},
cid = {I:(DE-2719)1210000},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:39820740},
doi = {10.1007/s11910-024-01400-8},
url = {https://pub.dzne.de/record/276089},
}