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@ARTICLE{Rudnicki:281786,
author = {Rudnicki, Stacy A and Al-Chalabi, Ammar and Andrews, Jinsy
A and Chio, Adriano and Corcia, Philippe and Couratier,
Philippe and Cudkowicz, Merit E and De Carvalho, Mamede and
Genge, Angela and Hardiman, Orla and Heiman-Patterson, Terry
and Henderson, Robert D and Ingre, Caroline and Johnston,
Wendy and Ludolph, Albert and Maragakis, Nicholas J and
Miller, Timothy M and Mora, Jesus S and Petri, Susanne and
Simmons, Zachary and Van Den Berg, Leonard H and Zinman,
Lorne and Herder, Katherine E and Kupfer, Stuart and Malik,
Fady I and Meng, Lisa and Simkins, Tyrell J and Wei, Jenny
and Wolff, Andrew A and Shefner, Jeremy M},
collaboration = {group, Courage-Als study},
title = {{H}ospitalizations as an outcome measure in
{COURAGE}-{ALS}.},
journal = {Amyotrophic lateral sclerosis $\&$ frontotemporal
degeneration},
volume = {26},
number = {7-8},
issn = {2167-8421},
address = {Abingdon},
publisher = {Taylor Francis Group},
reportid = {DZNE-2025-01179},
pages = {802 - 811},
year = {2025},
abstract = {Objective: To describe the development of a methodology to
characterize hospitalizations and their relationship to
amyotrophic lateral sclerosis (ALS) and provide results
using this process in a phase 3 trial of reldesemtiv in ALS.
Methods: ALS clinical trialists assisted in developing a
classification system to determine if a hospitalization was
related to ALS (HR-ALS), unrelated (HU-ALS), or if the
relationship was indeterminate (HI-ALS) and this was applied
by the investigators to hospitalizations in COURAGE-ALS.
Time to first hospitalization and number of hospitalizations
were compared between those assigned reldesemtiv or placebo
for up to 48 weeks. Demographic and clinical features were
evaluated for prediction of hospitalization risk; this
analysis was limited to those participants who completed the
first 24-week double-blind placebo-controlled portion of the
trial. Results: COURAGE-ALS terminated early due to
futility. Time to first hospitalization was similar in the
reldesemtiv compared to placebo arms as was the incidence,
with 86 of the participants $(17.6\%$ of those originally
assigned placebo and $18.0\%$ originally on reldesemtiv)
experiencing an event. The largest percentage of events was
classified as HR-ALS for both placebo $(64\%,$ 18/28) and
reldesemtiv $(76\%,$ 44/58). In a multivariate model, only
bulbar or respiratory onset disease was a significant risk
factor for hospitalization. Conclusion: While most
hospitalizations in COURAGE-ALS were HR-ALS, HU-ALS and
HI-ALS also occurred. When using hospitalization as an
endpoint in an ALS clinical trial, recording its
relationship to ALS provides additional details to
characterize disease burden and clinical meaningfulness of
the endpoint.},
keywords = {Humans / Amyotrophic Lateral Sclerosis: drug therapy /
Amyotrophic Lateral Sclerosis: therapy / Amyotrophic Lateral
Sclerosis: diagnosis / Amyotrophic Lateral Sclerosis:
epidemiology / Hospitalization: statistics $\&$ numerical
data / Male / Female / Middle Aged / Double-Blind Method /
Aged / Outcome Assessment, Health Care: methods /
Amyotrophic lateral sclerosis (Other) / COVID-19 (Other) /
fast skeletal muscle troponin activator (Other) /
hospitalization (Other) / motor neuron disease (Other) /
reldesemtiv (Other)},
cin = {Clinical Study Center (Ulm)},
ddc = {610},
cid = {I:(DE-2719)5000077},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40503807},
doi = {10.1080/21678421.2025.2515907},
url = {https://pub.dzne.de/record/281786},
}