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@ARTICLE{Indelicato:281817,
author = {Indelicato, Elisabetta and Fleszar, Zofia and Pellerin,
David and Nachbauer, Wolfgang and Zuchner, Stephan and
Traschütz, Andreas and Amprosi, Matthias and Schöls,
Ludger and Haack, Tobias B and Brais, Bernard and Boesch,
Sylvia and Synofzik, Matthis},
title = {{GAA}-{FGF}14 {E}xpansions and {CACNA}1{A} {V}ariants:
{P}henotypic {O}verlap and {D}iagnostic {I}mplications.},
journal = {Movement disorders},
volume = {40},
number = {10},
issn = {0885-3185},
address = {New York, NY},
publisher = {Wiley},
reportid = {DZNE-2025-01199},
pages = {2262 - 2268},
year = {2025},
abstract = {An intronic (GAA)•(TTC) repeat expansion in FGF14 was
recently identified as the cause of spinocerebellar ataxia
27B (SCA27B), a disorder presenting with both chronic
cerebellar ataxia and episodic symptoms. The phenotype of
SCA27B overlaps with that of CACNA1A spectrum disorders.The
objective of this work was to investigate the prevalence of
GAA-FGF14 repeat expansions in patients with ataxia so far
considered to be related to underlying CACNA1A variants.This
is a cross-sectional multicenter study.GAA-FGF14 testing
showed pathogenic expansions (≥250 repeats) in 6/67
$(9\%)$ patients carrying CACNA1A variants. All patients
with a pathogenic GAA-FGF14 expansion had a disease onset
>40 years and carried variants of uncertain significance
(VUSs) in CACNA1A. Genetic reevaluation led to the
reclassification of CACNA1A VUSs as likely benign in four of
six patients, who were ultimately diagnosed with
SCA27B.Late-onset ataxia cases previously considered as
CACNA1A-related disorder should be reevaluated and tested
for SCA27B, particularly if related to a VUS in CACNA1A. ©
2025 The Author(s). Movement Disorders published by Wiley
Periodicals LLC on behalf of International Parkinson and
Movement Disorder Society.},
keywords = {Humans / Male / Female / Middle Aged / Cross-Sectional
Studies / Adult / Fibroblast Growth Factors: genetics /
Phenotype / Aged / Calcium Channels: genetics /
Spinocerebellar Ataxias: genetics / Spinocerebellar Ataxias:
diagnosis / CACNA1A (Other) / GAA‐FGF14 ataxia (Other) /
SCA27B (Other) / episodic ataxia (Other) / spinocerebellar
ataxia 27B (Other) / CACNA1A protein, human (NLM Chemicals)
/ fibroblast growth factor 14 (NLM Chemicals) / Fibroblast
Growth Factors (NLM Chemicals) / Calcium Channels (NLM
Chemicals)},
cin = {AG Gasser / AG Schöls},
ddc = {610},
cid = {I:(DE-2719)1210000 / I:(DE-2719)5000005},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40879304},
doi = {10.1002/mds.30328},
url = {https://pub.dzne.de/record/281817},
}