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@ARTICLE{Calandrini:170414,
author = {C. Calandrini and S. R. van Hooff and I. Paassen and D.
Ayyildiz and S. Derakhshan and M. E. M. Dolman and K. P. S.
Langenberg and M. van de Ven and C. de Heus and N. Liv and
M. Kool$^*$ and R. R. de Krijger and G. A. M. Tytgat and M.
M. van den Heuvel-Eibrink and J. J. Molenaar and J. Drost},
title = {{O}rganoid-based drug screening reveals neddylation as
therapeutic target for malignant rhabdoid tumors.},
journal = {Cell reports},
volume = {36},
number = {8},
issn = {2211-1247},
address = {[New York, NY]},
publisher = {Elsevier},
reportid = {DKFZ-2021-01917},
pages = {109568},
year = {2021},
abstract = {Malignant rhabdoid tumors (MRTs) represent one of the most
aggressive childhood malignancies. No effective treatment
options are available, and prognosis is, therefore, dismal.
Previous studies have demonstrated that tumor organoids
capture the heterogeneity of patient tumors and can be used
to predict patient response to therapy. Here, we perform
drug screening on patient-derived normal and tumor organoids
to identify MRT-specific therapeutic vulnerabilities. We
identify neddylation inhibitor MLN4924 as a potential
therapeutic agent. Mechanistically, we find increased
neddylation in MRT organoids and tissues and show that
MLN4924 induces a cytotoxic response via upregulation of the
unfolded protein response. Lastly, we demonstrate in vivo
efficacy in an MRT PDX mouse model, in which single-agent
MLN4924 treatment significantly extends survival. Our study
demonstrates that organoids can be used to find drugs
selectively targeting tumor cells while leaving healthy
cells unharmed and proposes neddylation inhibition as a
therapeutic strategy in MRT.},
keywords = {MLN4924 (Other) / drug screening (Other) / malignant
rhabdoid tumors (Other) / neddylation (Other) / organoids
(Other) / targeted therapy (Other)},
cin = {B062 / HD01},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)HD01-20160331},
pnm = {312 - Funktionelle und strukturelle Genomforschung
(POF4-312)},
pid = {G:(DE-HGF)POF4-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:34433038},
doi = {10.1016/j.celrep.2021.109568},
url = {https://inrepo02.dkfz.de/record/170414},
}