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@ARTICLE{Ziegler:302116,
author = {M. Ziegler$^*$ and N. Khoury$^*$ and L. M. Hommerich$^*$
and H. Adler$^*$ and S. Loges$^*$},
title = {{F}unctional {C}haracterization of {V}ariants of {U}nknown
{S}ignificance of {F}ibroblast {G}rowth {F}actor {R}eceptors
1-4 and {C}omparison {W}ith {AI} {M}odel-{B}ased
{P}rediction.},
journal = {JCO precision oncology},
volume = {9},
number = {9},
issn = {2473-4284},
address = {Alexandria, VA},
publisher = {American Society of Clinical Oncology},
reportid = {DKFZ-2025-01248},
pages = {e2400847},
year = {2025},
note = {#EA:A420#LA:A420#},
abstract = {Fibroblast growth factor receptors (FGFRs; FGFR1, FGFR2,
FGFR3, FGFR4) are frequently mutated oncogenes in solid
cancers. The oncogenic potential of FGFR rearrangements and
few hotspot point mutations is well established, but the
majority of variants resulting from point mutations
especially outside of the tyrosine kinase domain are
currently considered variants of unknown significance
(VUS).Recurrent nonkinase domain FGFR VUS variants were
collected from the Catalog of Somatic Mutations in Cancer
and their oncogenic potential was assessed in vitro by
different functional assays. We compiled published clinical
and preclinical data on FGFR variants and compared the data
with results from our functional assays and pathogenicity
predictions of state-of-the-art artificial intelligence (AI)
models.We identified 12 novel FGFR extracellular small
variants with potential driver function. Comparison of
clinical and preclinical data on FGFR variants with
pathogenicity predictions of state-of-the-art AI models
showed limited usefulness of the AI predictions. Sensitivity
profiles of activating FGFR variants to targeted inhibitors
were recorded and showed good targetability of FGFR
nonkinase domain variants.The collected results extend the
spectrum of suitable FGFR variants for potential treatment
with FGFR inhibitors in the context of clinical trials and
beyond. Current AI models for variant pathogenicity
prediction require further refinement for use in oncogenic
decision making.},
keywords = {Humans / Artificial Intelligence / Neoplasms: genetics /
Mutation / Receptors, Fibroblast Growth Factor: genetics /
Receptors, Fibroblast Growth Factor (NLM Chemicals)},
cin = {A420},
ddc = {610},
cid = {I:(DE-He78)A420-20160331},
pnm = {311 - Zellbiologie und Tumorbiologie (POF4-311)},
pid = {G:(DE-HGF)POF4-311},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40526877},
doi = {10.1200/PO-24-00847},
url = {https://inrepo02.dkfz.de/record/302116},
}