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@ARTICLE{Pauck:298952,
author = {D. Pauck$^*$ and D. J. Picard$^*$ and M. Maue$^*$ and K.
Taban$^*$ and V. Marquardt$^*$ and L. Blümel$^*$ and J.
Bartl$^*$ and N. Qin and N. Kubon$^*$ and D. Schöndorf and
F.-D. Meyer$^*$ and J. Theruvath and S. Mitra and M.
Hasselblatt and M. C. Frühwald and G. Reifenberger$^*$ and
M. Remke$^*$},
title = {{A}n in vitro pharmacogenomic approach reveals
subtype-specific therapeutic vulnerabilities in atypical
teratoid/rhabdoid tumors ({AT}/{RT}).},
journal = {Pharmacological research},
volume = {213},
issn = {1043-6618},
address = {London},
publisher = {Academic Press},
reportid = {DKFZ-2025-00386},
pages = {107660},
year = {2025},
note = {Volume 213, March 2025, 107660},
abstract = {Atypical teratoid/rhabdoid tumor (AT/RT) is a highly
malignant embryonal brain tumor driven by genetic
alterations inactivating the SMARCB1 or, less commonly, the
SMARCA4 gene. Large-scale molecular profiling studies have
identified distinct molecular subtypes termed AT/RT-TYR,
-SHH and -MYC. Despite the increasing knowledge of AT/RT
biology, curative treatment options are still lacking for
certain risk groups and outcomes of these patients remain
poor. We performed an in vitro high-throughput drug screen
of 768 small molecule drugs covering conventional
chemotherapeutic agents and late-stage developmental drugs
in 13 AT/RT cell lines and determined intra- and
inter-entity differential responses to unravel specific
vulnerabilities. Our data demonstrated in vitro preferential
activity of mitogen-activated protein kinase kinase (MEK)
and mouse double minute 2 homolog (MDM2) inhibitors in AT/RT
cell lines compared to other high-grade brain tumor cell
lines including medulloblastoma and malignant glioma models.
Moreover, we were able to link distinct drug response
patterns to AT/RT molecular subtypes through integration of
drug response data with large-scale DNA methylation and
RNASeq-based expression profiles. Subtype-dependent drug
response profiles demonstrated sensitivity of AT/RT-SHH cell
lines to B-cell lymphoma 2 (BCL2) and heat shock protein 90
(HSP90) inhibitors, and increased activity of microtubule
inhibitors, kinesin spindle protein (KSP) inhibitors, and
the eukaryotic translation initiation factor 4E (eIF4E)
inhibitor briciclib in a subset of AT/RT-MYC cell lines. In
summary, our in vitro pharmacogenomic approach revealed
preclinical evidence of tumor type- and subtype-specific
therapeutic vulnerabilities in AT/RT cell lines that may
inform future in vivo and clinical evaluations of novel
pharmacological strategies.},
keywords = {17-AAG/Tanespimycin (PubChem CID: 6505803) (Other) /
ABT-199/Venetoclax (PubChem CID: 49846579) (Other) / ABT-737
(PubChem CID: 11228183) (Other) / Atypical teratoid/rhabdoid
tumor (Other) / DNA methylation profiling (Other) / GDC-0623
(PubChem CID: 42642654) (Other) / RNA sequencing (Other) /
briciclib (PubChem CID: 11248490) (Other) / high-throughput
drug screening (Other) / idasanutlin (PubChem CID: 53358942)
(Other) / ombrabulin hydrochloride (PubChem CID: 6918404)
(Other) / targeted therapy (Other)},
cin = {ED01},
ddc = {610},
cid = {I:(DE-He78)ED01-20160331},
pnm = {899 - ohne Topic (POF4-899)},
pid = {G:(DE-HGF)POF4-899},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:39961404},
doi = {10.1016/j.phrs.2025.107660},
url = {https://inrepo02.dkfz.de/record/298952},
}