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@ARTICLE{Hglinger:139314,
author = {Höglinger, Günter U and Schöpe, Jakob and Stamelou,
Maria and Kassubek, Jan and Del Ser, Teodoro and Boxer, Adam
L and Wagenpfeil, Stefan and Huppertz, Hans-Jürgen and
Investigators, AL-108-231 and Investigators, Tauros MRI and
Group, Movement Disorder Society-Endorsed PSP Study},
title = {{L}ongitudinal magnetic resonance imaging in progressive
supranuclear palsy: {A} new combined score for clinical
trials.},
journal = {Movement disorders},
volume = {32},
number = {6},
issn = {0885-3185},
address = {New York, NY},
publisher = {Wiley},
reportid = {DZNE-2020-05636},
pages = {842-852},
year = {2017},
abstract = {Two recent, randomized, placebo-controlled phase II/III
trials (clinicaltrials.gov: NCT01110720, NCT01049399) of
davunetide and tideglusib in progressive supranuclear palsy
(PSP) generated prospective, 1-year longitudinal datasets of
high-resolution T1-weighted three-dimensional MRI.The
objective of this study was to develop a quantitative MRI
disease progression measurement for clinical trials.The
authors performed a fully automated quantitative MRI
analysis employing atlas-based volumetry and provide sample
size calculations based on data collected in 99 PSP patients
assigned to placebo in these trials. Based on individual
volumes of 44 brain compartments and structures at baseline
and 52 weeks of follow-up, means and standard deviations of
annualized percentage volume changes were used to estimate
standardized effect sizes and the required sample sizes per
group for future 2-armed, placebo-controlled therapeutic
trials.The highest standardized effect sizes were found for
midbrain, frontal lobes, and the third ventricle. Using the
annualized percentage volume change of these structures to
detect a $50\%$ change in the 1-year progression $(80\%$
power, significance level $5\%)$ required lower numbers of
patients per group (third ventricle, n = 32; midbrain, n =
37; frontal lobe, n = 43) than the best clinical scale (PSP
rating scale total score, n = 58). A combination of volume
changes in these 3 structures reduced the number of required
patients to only 20 and correlated best with the progression
in the clinical scales.We propose the 1-year change in the
volumes of third ventricle, midbrain, and frontal lobe as
combined imaging read-out for clinical trials in PSP that
require the least number of patients for detecting efficacy
to reduce brain atrophy. © 2017 International Parkinson and
Movement Disorder Society.},
keywords = {Aged / Clinical Trials as Topic: methods / Disease
Progression / Female / Frontal Lobe: diagnostic imaging /
Humans / Longitudinal Studies / Magnetic Resonance Imaging:
methods / Male / Mesencephalon: diagnostic imaging / Middle
Aged / Supranuclear Palsy, Progressive: diagnostic imaging /
Third Ventricle: diagnostic imaging},
cin = {AG Höglinger 1},
ddc = {610},
cid = {I:(DE-2719)1110002},
pnm = {344 - Clinical and Health Care Research (POF3-344)},
pid = {G:(DE-HGF)POF3-344},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:28436538},
pmc = {pmc:PMC5808453},
doi = {10.1002/mds.26973},
url = {https://pub.dzne.de/record/139314},
}