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Record W7135033096 · doi:10.1093/neuped/wuaf001.071

DMG-41. Development of Brain-Penetrant ALK2 Inhibitors for Diffuse Intrinsic Pontine Glioma (DIPG): An Open Science Approach.

2025· article· en· W7135033096 on OpenAlex
Peter B. Sampson, David Smil, Hector Gonzales-Alvarez, Deeba Ensan, Max Morgan, Taira Kiyota, Carlos Zepeda-Velazquez, Ahmed Aman, Methvin Isaac, Diana Carvahlo, Jiin Song, Rebecca Rogers, Chris Jones, Angel Montero Carcaboso, Oren J. Becher, David Uehling, R. Al-Awar

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

affAt least one author lists a Canadian institution in the pinned OpenAlex snapshot.

Bibliographic record

VenueNeuro-Oncology Pediatrics · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicTGF-β signaling in diseases
Canadian institutionsUniversity of TorontoOntario Institute for Cancer ResearchFSD Pharma (Canada)
Fundersnot available
KeywordsGliomaMutantMissense mutationDrug discoveryPonsIn vivo

Abstract

fetched live from OpenAlex

Abstract Diffuse Intrinsic Pontine Glioma (DIPG) is a rare and aggressive pediatric cancer located in the pons region of the brainstem, classified as part of a broader class of H3 K27M mutant Diffuse Midline Gliomas (DMG). Focal radiation remains the standard of care; however the median survival is only 9-12 months, with no effective chemotherapeutic options available. Somatic missense mutations in the bone morphogenetic protein (BMP) type I receptor ACVR1 gene, which encodes activin receptor-like kinase-2 (ALK2), are present in approximately 25% of DMG tumours. Using an Open Science approach to promote transparency and collaboration, we previously reported ALK2 inhibitors with high potency (ALK2 IC50 < 10 nM, nanoBRET ALK2 IC50 < 20 nM), selectivity (>100-fold over ALK5) and brain penetration (Cbrain,4h/Cplasma,4h > 0.5, 10 mg/kg, NOD-SCID male mice). These inhibitors are highly orally bioavailable and demonstrate high in vivo exposure and dose linearity. In vitro, compounds have been screened in a panel of patient-derived DMG models including ACVR1 mutant and wild-type, with GI50s ranging from 0.3–14 µM, and selectivity for mutant over wild-type. In vivo, compounds showed an extension of survival in both an orthotopic PDX model (HSJD-DIPG-007, H3.3_K27M, ACVR1_R206H), and in an allograft of RCAS-ACVR1_R206H/H3.1_K27M in an immune competent background. From this series, we describe the selection process for a clinical development candidate. This work highlights the potential of ALK2 inhibitors as a therapeutic strategy for ACVR1 mutant gliomas and the value of Open Science in accelerating drug discovery for rare diseases.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.002
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.094
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0020.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.017
GPT teacher head0.306
Teacher spread0.289 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it