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@ARTICLE{Gatz:307659,
author = {S. A. Gatz and J. Glade-Bender and A. D. J. Pearson and M.
V. Ortiz and R. Bernardi and L. Chesler and S. Clifford and
S. Cohen-Gogo and E. De La Cuesta and T. de Rojas and K.
Durinck and S. Federico and E. Fox and S. George and I.
Gounaris and A. G. Henssen and M. Irwin and M. Kool$^*$ and
A. Lau and K. Nysom and A. Pappo and G. K. Pennock and S. M.
Pfister$^*$ and N. Scobie and E. K. Slotkin and M. Smith and
F. Speleman and E. A. Stewart and B. J. Weigel and G.
Vassal},
title = {{P}athway for the {D}evelopment of {ATR} {I}nhibitors in
{P}ediatric {M}alignancies: {A}n {ACCELERATE}
{M}ultistakeholder {A}nalysis.},
journal = {JCO precision oncology},
volume = {10},
number = {10},
issn = {2473-4284},
address = {Alexandria, VA},
publisher = {American Society of Clinical Oncology},
reportid = {DKFZ-2026-00116},
pages = {e2500642},
year = {2026},
note = {#NCTZFB26# / #DKTKZFB26#},
abstract = {High levels of DNA replication stress and defects in the
DNA damage response (DDR) pathways are vulnerabilities of
many poor prognosis childhood malignancies. Ataxia
telangiectasia and Rad3-related protein (ATR) is a key
regulator of these pathways and constitutes an attractive
target, especially in combination. However, the malignancies
where ATR inhibitors have maximum benefit and synergistic
combinations differ between adults and children.ACCELERATE
convened a multistakeholder meeting and conducted review and
analysis to propose the optimal pathway for the development
of ATR inhibitors in pediatric malignancies.Considering the
lack of identified biomarkers, the initial evaluation of ATR
inhibitors should focus on Ewing sarcoma, rhabdomyosarcoma,
and neuroblastoma in view of their high levels of DNA
replication stress and defects in DDR pathways. Early phase
trials of ATR inhibitors should be iterative, based on a
clear hypothesis with responders and nonresponders
undergoing detailed molecular analysis and a revised new
hypothesis generated. Trial designs should restrict
monotherapy evaluation to a brief exposure in a small number
of patients and progress rapidly to combinations.
Highlighted combination partners are poly(ADP-ribose)
polymerase inhibitors and antibody drug conjugates with
topoisomerase I inhibitor payloads. Combinations with ALK
inhibitors (in ALK/MYCN-aberrant neuroblastoma) and aurora A
kinase (in MYCN-amplified) are supported by robust
mechanisms of action and preclinical data. Early
interactions with regulators are crucial, and early phase
clinical trials should be conducted in regulatory-approved,
academic-sponsored, industry-supported, platform trials.ATR
inhibitors are a prototype for the development of medicinal
products in a limited pediatric population. For the
substantial potential of ATR inhibitors in children with
malignancy to be realized, strategic planning between
academia, industry, regulators, and patient advocates is
vital.},
subtyp = {Review Article},
keywords = {Humans / Ataxia Telangiectasia Mutated Proteins:
antagonists $\&$ inhibitors / Child / Neoplasms: drug
therapy / Neuroblastoma: drug therapy / Sarcoma, Ewing: drug
therapy / Protein Kinase Inhibitors: therapeutic use /
Protein Kinase Inhibitors: pharmacology / Ataxia
Telangiectasia Mutated Proteins (NLM Chemicals) / ATR
protein, human (NLM Chemicals) / Protein Kinase Inhibitors
(NLM Chemicals)},
cin = {B062 / HD01 / HD02},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)HD01-20160331 /
I:(DE-He78)HD02-20160331},
pnm = {312 - Funktionelle und strukturelle Genomforschung
(POF4-312)},
pid = {G:(DE-HGF)POF4-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:41538758},
doi = {10.1200/PO-25-00642},
url = {https://inrepo02.dkfz.de/record/307659},
}