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@ARTICLE{Maas:285346,
author = {Maas, Roderick P P W M and Garcia-Moreno, Hector and Faber,
Jennifer and Gonzalez, Carlos and Schöls, Ludger and de
Vries, Jeroen J and Bushara, Khalaf and Reetz, Kathrin and
Onyike, Chiadi U and Jacobi, Heike and Erdlenbruch,
Friedrich and Infante, Jon and Santana, Magda M and
Hübener-Schmid, Jeannette and de Almeida, Luís Pereira and
Lima, Manuela and Giunti, Paola and Klockgether, Thomas and
van de Warrenburg, Bart P C},
collaboration = {group, ESMI study},
title = {{C}ognitive impairment in {SCA}3: {A} multi-center cohort
study with demographic, imaging, and biomarker correlates.},
journal = {Neurobiology of disease},
volume = {220},
issn = {0969-9961},
address = {[Amsterdam]},
publisher = {Elsevier},
reportid = {DZNE-2026-00212},
pages = {107301},
year = {2026},
abstract = {Cognitive deficits are common in spinocerebellar ataxia
type 3 (SCA3), but their neurobiological correlates remain
largely unknown.To investigate cognitive performance in a
large international cohort of SCA3 mutation carriers
covering the entire disease course and to explore
associations with posterior cerebellar volumes, basal
ganglia and thalamus volumes, and plasma neurofilament light
chain (NfL) concentration.The Montreal Cognitive Assessment
(MoCA) was used to evaluate cognitive impairment in this
prospective, observational cohort study involving 13 ataxia
referral centers. Standardized motor assessments, brain MR
imaging, and peripheral blood biosampling were also
performed.MoCA data were collected from 61 pre-ataxic SCA3
mutation carriers, 231 ataxic SCA3 patients, and 111 healthy
controls. After adjustments for educational level and age,
there were significant differences in MoCA total score, as
well as visuospatial/executive, attention, language, and
abstraction subscores, between healthy controls and ataxic,
but not pre-ataxic individuals. MoCA scores declined with
ataxia severity, especially in patients with a lower
educational level. Patients with a MoCA score < 26 had lower
pallidal volumes and higher plasma NfL concentrations than
those with a score ≥ 26. However, only the interaction
term between ataxia severity and educational level was
independently associated with cognitive performance in
multivariable regression analyses containing demographic,
clinical, volumetric, and biochemical parameters.Cognitive
deficits in SCA3 generally appear after clinical ataxia
onset and progress in parallel with ataxia severity,
especially in patients with a lower cognitive reserve. Other
measured biochemical and imaging parameters did not have a
significant additional contribution.},
keywords = {Humans / Male / Female / Middle Aged / Cognitive
Dysfunction: diagnostic imaging / Cognitive Dysfunction:
etiology / Cognitive Dysfunction: blood / Cognitive
Dysfunction: psychology / Adult / Biomarkers: blood /
Magnetic Resonance Imaging / Machado-Joseph Disease:
complications / Machado-Joseph Disease: diagnostic imaging /
Machado-Joseph Disease: psychology / Machado-Joseph Disease:
genetics / Machado-Joseph Disease: blood / Cohort Studies /
Neurofilament Proteins: blood / Aged / Prospective Studies /
Mental Status and Dementia Tests / Cerebellum (Other) /
Cognition (Other) / Machado-Joseph disease (Other) /
Spinocerebellar ataxia type 3 (Other) / Biomarkers (NLM
Chemicals) / Neurofilament Proteins (NLM Chemicals) /
neurofilament protein L (NLM Chemicals)},
cin = {Clinical Research (Bonn) / AG Schöls / Patient Studies
(Bonn)},
ddc = {570},
cid = {I:(DE-2719)1011001 / I:(DE-2719)5000005 /
I:(DE-2719)1011101},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
experiment = {EXP:(DE-2719)ESMI-20140101},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:41654200},
doi = {10.1016/j.nbd.2026.107301},
url = {https://pub.dzne.de/record/285346},
}