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@ARTICLE{Umbaugh:304303,
author = {C. S. Umbaugh$^*$ and M. Groth$^*$ and C. Erkut$^*$ and K.
Lee$^*$ and J. Marinho and S. Linder$^*$ and F. Iser$^*$ and
J. Kapp$^*$ and P. Schroeter$^*$ and S. Dolaner$^*$ and A.
Kayserili$^*$ and D. Helm$^*$ and M. Schneider$^*$ and J.
Hartmann$^*$ and P. Walch$^*$ and T. F. E. Barth and K.
Mellert and B. Dreier and J. V. Schaefer and A. Plückthun
and S. Fröhling$^*$ and C. Scholl$^*$},
title = {{S}elective targeting of {TBXT} with {DARP}ins identifies
regulatory networks and therapeutic vulnerabilities in
chordoma.},
journal = {Science advances},
volume = {11},
number = {36},
issn = {2375-2548},
address = {Washington, DC [u.a.]},
publisher = {Assoc.},
reportid = {DKFZ-2025-01838},
pages = {eadu2796},
year = {2025},
note = {#EA:B290#EA:B340#LA:B290#LA:B340#},
abstract = {The embryonic transcription factor TBXT (brachyury) drives
chordoma, a spinal neoplasm without effective drug
therapies. TBXT's regulatory network is poorly understood,
and strategies to disrupt its activity for therapeutic
purposes are lacking. We developed designed ankyrin repeat
proteins that block TBXT-DNA binding (T-DARPins). In
chordoma cells, T-DARPins reduced cell cycle progression,
spheroid formation, and tumor growth in mice and induced
signs of senescence and differentiation. Transcriptomic and
proteomic analyses identified gene networks involved in cell
cycle regulation, embryonic cell identity, and interferon
response and revealed features of regulome components, such
as susceptibility to pharmacologic inhibition and the
fine-tuning of TBXT downstream effectors through IGFBP3.
Finally, we found high interferon signaling in chordoma cell
lines and patient tumors, which was promoted by TBXT and
associated with sensitivity to JAK2 inhibitors. These
findings demonstrate the potential of DARPins for probing
nuclear proteins to understand the regulatory networks of
transcription factor-driven cancers, including entry points
for therapies that warrant testing in patients.},
keywords = {Humans / Animals / Gene Regulatory Networks / Mice /
Chordoma: metabolism / Chordoma: genetics / Chordoma: drug
therapy / Chordoma: pathology / Cell Line, Tumor / Gene
Expression Regulation, Neoplastic: drug effects / Proteomics
/ Xenograft Model Antitumor Assays},
cin = {B290 / B340 / HD01 / W120},
ddc = {500},
cid = {I:(DE-He78)B290-20160331 / I:(DE-He78)B340-20160331 /
I:(DE-He78)HD01-20160331 / I:(DE-He78)W120-20160331},
pnm = {312 - Funktionelle und strukturelle Genomforschung
(POF4-312)},
pid = {G:(DE-HGF)POF4-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40901948},
doi = {10.1126/sciadv.adu2796},
url = {https://inrepo02.dkfz.de/record/304303},
}