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@ARTICLE{Kasper:1002271,
author = {Kasper, Jan and Eickhoff, Simon B and Caspers, Svenja and
Peter, Jessica and Dogan, Imis and Wolf, Robert Christian
and Reetz, Kathrin and Dukart, Juergen and Orth, Michael},
title = {{L}ocal synchronicity in dopamine-rich caudate nucleus
influences {H}untington’s disease motor phenotype},
journal = {Brain},
volume = {146},
number = {8},
issn = {0006-8950},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {FZJ-2023-01253},
pages = {3319–3330},
year = {2023},
abstract = {Structural grey and white matter changes precede the
manifestation of clinical signs of Huntington's disease by
many years. Conversion to clinically manifest disease
therefore likely reflects not merely atrophy but a more
wide-spread breakdown of brain function. Here, we
investigated the structure - function relationship close to
and after the clinical onset testing for its co-localization
with specific neurotransmitter/receptor systems and
important regional brain hubs, in particular caudate nucleus
and putamen that are central to maintain normal motor
behavior. In two independent cohorts of patients with
premanifest Huntington's disease close to onset and very
early manifest Huntington's disease (total n = 84; n = 88
matched controls) we used structural and resting state
functional MRI. We show that measures of functional activity
and local synchronicity within cortical and subcortical
regions remain normal in the premanifest Huntington's
disease phase despite clear evidence of brain atrophy. In
manifest Huntington's disease, homeostasis of synchronicity
was disrupted in subcortical hub regions such as caudate
nucleus and putamen, but also in cortical hub regions, for
instance the parietal lobe. Cross-modal spatial correlations
of functional MRI data with receptor/neurotransmitter
distribution maps showed that Huntington's disease specific
alterations co-localize with dopamine receptors D1, D2, as
well as dopamine and serotonin transporters. Caudate nucleus
synchronicity significantly improved models predicting the
severity of the motor phenotype or predicting the
classification into premanifest Huntington's disease or
motor manifest Huntington's disease. Our data suggest that
the functional integrity of the dopamine receptor rich
caudate nucleus is key to maintain network function. The
loss of caudate nucleus functional integrity affects network
function to a degree that causes a clinical phenotype. These
insights into what happens in Huntington's disease could
serve as a model for what might be a more general
relationship between brain structure and function in
neurodegenerative diseases in which other brain regions are
vulnerable.},
cin = {INM-7 / INM-1 / INM-11},
ddc = {610},
cid = {I:(DE-Juel1)INM-7-20090406 / I:(DE-Juel1)INM-1-20090406 /
I:(DE-Juel1)INM-11-20170113},
pnm = {5251 - Multilevel Brain Organization and Variability
(POF4-525) / HBP SGA3 - Human Brain Project Specific Grant
Agreement 3 (945539)},
pid = {G:(DE-HGF)POF4-5251 / G:(EU-Grant)945539},
typ = {PUB:(DE-HGF)16},
pubmed = {36795496},
UT = {WOS:000954320200001},
doi = {10.1093/brain/awad043},
url = {https://juser.fz-juelich.de/record/1002271},
}