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@ARTICLE{Grosch:888307,
author = {Grosch, Anne Sophie and Rinnenthal, Jan Leo and
Rönnefarth, Maria and Lux, Silke and Scheel, Michael and
Endres, Matthias and Brandt, Alexander U. and Paul,
Friedemann and Schmitz-Hübsch, Tanja and Minnerop, Martina
and Doss, Sarah},
title = {{N}eurochemical {D}ifferences in {S}pinocerebellar {A}taxia
{T}ype 14 and 1},
journal = {The Cerebellum},
volume = {20},
issn = {1473-4230},
address = {London},
publisher = {Dunitz},
reportid = {FZJ-2020-04827},
pages = {169–178},
year = {2021},
abstract = {Autosomal-dominant spinocerebellar ataxias (SCA) are
neurodegenerative diseases characterized by progressive
ataxia. Here, we report on neurometabolic alterations in
spinocerebellar ataxia type 1 (SCA1; SCA-ATXN1) and 14
(SCA14; SCA-PRKCG) assessed by non-invasive 1H magnetic
resonance spectroscopy. Three Tesla 1H magnetic resonance
spectroscopy was performed in 17 SCA14, 14 SCA1 patients,
and in 31 healthy volunteers. We assessed metabolites in the
cerebellar vermis, right cerebellar hemisphere, pons,
prefrontal, and motor cortex. Additionally, clinical
characteristics were obtained for each patient to correlate
them with metabolites. In SCA14, metabolic changes were
restricted to the cerebellar vermis compared with widespread
neurochemical alterations in SCA1. In SCA14, total
N-acetylaspartate (tNAA) was reduced in the vermis by
$34\%.$ In SCA1, tNAA was reduced in the vermis $(24\%),$
cerebellar hemisphere $(26\%),$ and pons $(25\%).$ SCA14
patients showed $24\%$ lower glutamate+glutamine (Glx) and
$46\%$ lower γ-aminobutyric acid (GABA) in the vermis,
while SCA1 patients showed no alterations in Glx and GABA.
SCA1 revealed a decrease of aspartate (Asp) in the vermis
$(62\%)$ and an elevation in the prefrontal cortex $(130\%)$
as well as an elevation of myo-inositol (Ins) in the
cerebellar hemisphere $(51\%)$ and pons $(46\%).$ No changes
of Asp and Ins were detected in SCA14. Beyond, glucose (Glc)
was increased in the vermis of both SCA14 $(155\%)$ and SCA1
$(247\%).$ 1H magnetic resonance spectroscopy revealed
differing neurochemical profiles in SCA1 and SCA14 and
confirmed metabolic changes that may be indicative for
neuronal loss and dysfunctional energy metabolism.
Therefore, 1H magnetic resonance spectroscopy represents a
helpful tool for in-vivo tracking of disease-specific
pathophysiology.},
cin = {INM-1},
ddc = {610},
cid = {I:(DE-Juel1)INM-1-20090406},
pnm = {5251 - Multilevel Brain Organization and Variability
(POF4-525)},
pid = {G:(DE-HGF)POF4-5251},
typ = {PUB:(DE-HGF)16},
pubmed = {33063293},
UT = {WOS:000578890100001},
doi = {10.1007/s12311-020-01201-y},
url = {https://juser.fz-juelich.de/record/888307},
}