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@ARTICLE{Schmidt:131608,
author = {C. Schmidt$^*$ and N. A. Schubert and S. Brabetz$^*$ and N.
Mack$^*$ and B. Schwalm$^*$ and J. A. Chan and F. Selt$^*$
and C. Herold-Mende and O. Witt$^*$ and T. Milde$^*$ and S.
Pfister$^*$ and A. Korshunov$^*$ and M. Kool$^*$},
title = {{P}reclinical drug screen reveals topotecan, actinomycin
{D}, and volasertib as potential new therapeutic candidates
for {ETMR} brain tumor patients.},
journal = {Neuro-Oncology},
volume = {19},
number = {12},
issn = {1523-5866},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {DKFZ-2017-06240},
pages = {1607 - 1617},
year = {2017},
abstract = {Embryonal tumor with multilayered rosettes (ETMR) is a rare
and aggressive embryonal brain tumor that solely occurs in
infants and young children and has only recently been
recognized as a separate brain tumor entity in the World
Health Organization classification for CNS tumors. Patients
have a very dismal prognosis with a median survival of 12
months upon diagnosis despite aggressive treatment. The aim
of this study was to develop novel treatment regimens in a
preclinical drug screen in order to inform potentially more
active clinical trial protocols.We have carried out an in
vitro and in vivo drug screen using the ETMR cell line BT183
and its xenograft model. Furthermore, we have generated the
first patient-derived xenograft (PDX) model for ETMR and
evaluated our top drug candidates in an in vitro drug screen
using this model.BT183 cells are very sensitive to the
topoisomerase inhibitors topotecan and doxorubicin, to the
epigenetic agents decitabine and panobinostat, to
actinomycin D, and to targeted drugs such as the polo-like
kinase 1 (PLK1) inhibitor volasertib, the aurora kinase A
inhibitor alisertib, and the mammalian target of rapamycin
(mTOR) inhibitor MLN0128. In xenograft mice, monotherapy
with topotecan, volasertib, and actinomycin D led to a
temporary response in tumor growth and a significant
increase in survival. Finally, using multi-agent treatment
regimens of topotecan or doxorubicin combined with
methotrexate and vincristine, the response in tumor growth
and survival was further increased compared with mice
receiving single treatments.We have identified several
promising candidates for combination therapies in future
clinical trials for ETMR patients.},
cin = {G340 / B062 / G380},
ddc = {610},
cid = {I:(DE-He78)G340-20160331 / I:(DE-He78)B062-20160331 /
I:(DE-He78)G380-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:28482026},
pmc = {pmc:PMC5716199},
doi = {10.1093/neuonc/nox093},
url = {https://inrepo02.dkfz.de/record/131608},
}