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Record W7135010552 · doi:10.1093/neuped/wuaf001.147

HGG-12. Integrated molecular profiling of diffuse midline glioma, H3K27-altered

2025· article· en· W7135010552 on OpenAlex
Yura Grabovska, Alan Mackay, Molina Das, Ketty Kessler, Anna Burford, Diana Carvalho, Rita Pereira, Laura Bevington, Sara Temelso, Drenusha Sejdiu, Valeria Molinari, Rebecca Rogers, Lynn Bjerke, Leslie Bridges, Zita Reisz, Prof Safa Al-Sarraj, Navneet Singh, Simon Stapleton, Cristina Bleil, Samantha Hettige, Bassel Zebian, Julia Cockle, Fernando Carceller, Matthew Clarke, Chris Jones

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
FieldMedicine
TopicGlioma Diagnosis and Treatment
Canadian institutionsInstitute of Cancer Research
Fundersnot available
KeywordsMethylationDNA methylationGliomaMAPK/ERK pathwayGene expression profilingMutationPhenotype

Abstract

fetched live from OpenAlex

Abstract Background Diffuse midline glioma, H3K27-altered (DMG-H3K27) are recognised as a distinct type of paediatric diffuse high-grade glioma with dismal clinical outcome in the WHO2021 CNS classification, defined by an integrated diagnosis including the defining oncohistone H3 alterations. Increasing evidence suggests the presence of distinct subtypes, but these are not yet well-defined. Methods Published and unpublished DNA sequencing from n = 554 DMG-H3K27 cases were integrated with n = 498 cases with methylation array profiling, along with bulk (n = 112) and single-cell (n = 30) RNAseq data. Results Clustering of methylation data by tSNE/UMAP showed a clear separation largely driven by anatomical location, with a cluster enriched for pontine lesions (DIPG, n = 225) separating from those enriched in non-brainstem lesions such as the thalamus (n = 258), tightly linked to H3 variant status, and highly enriched for gain of chromosome 1q or loss of 5q, respectively. The two major methylation-based clusters were readily discriminated on the basis of differentially methylated regions driving differential expression and scRNA-seq, highlighting the previously reported developmental origins associated with location, but also novel markers associated with differences in tumour immune microenvironment, and distinct subpopulations of cells driving tumour infiltration. Co-segregating mutations in the MAPK pathway, incorporating variously BRAF_V600E, NF1, FGFR1, and PTPN11 may define a clinically distinct group of tumours in the thalamus, though PIK3R1 InDels and non-canonical BRAF mutations in the pontine DMGs highlighted aggressive tumours for which in vitro modelling reveals initial sensitivity but also complex resistance to targeted MAPK inhibition. These tumours were clearly distinct from those of the DMG-EGFR subgroup, which clustered by methylation with diffusely infiltrating H3-WT hemispheric glioma, harboured frequent chromosome 7 gains absent from other DMG subtypes, hypomethylation of the EGFR/IGF1R loci, and EZHIP overexpression. Conclusion DMG-H3K27 comprise multiple subgroups with clinically relevant phenotypes driven by molecular features which arise within the context of distinct anatomical locations.

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)0.000
metaresearch head score (Gemma)0.001
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 score0.256
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
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 head0.015
GPT teacher head0.296
Teacher spread0.281 · 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