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Record W7135051555 · doi:10.1093/neuped/wuaf001.146

HGG-11. Integrated molecular profiling of H3 wild-type diffuse paediatric-type high-grade glioma

2025· article· en· W7135051555 on OpenAlex
Alan Mackay, Yura Grabovska, Rita Pereira, Anna Burford, Diana Carvalho, Sara Temelso, Drenusha Sejdiu, Shauna Crampsie, Laura Bevington, Valeria Molinari, Rebecca Rogers, Ketty Kessler, 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
KeywordsGliomaMethylationOligodendrogliomaPhenotypeDNA methylationDNAIncidence (geometry)

Abstract

fetched live from OpenAlex

Abstract Background Within the WHO2021 CNS Tumour Classification, oncohistone H3-mutations define around half of paediatric-type diffuse high grade glioma, however the remaining tumours (H3-WT-PDHGG) are less well described. Methods Published and unpublished DNA sequencing from n = 1601 H3-WT-PDHGG cases were integrated with n = 1847 cases with methylation array profiling, along with bulk (n = 251) and single-cell (n = 65) RNAseq data. Results Within H3-WT high-grade glioma, a total of 11 MNP12.8-defined subgroups were found to have a peak incidence <18years, excluding infant hemispheric glioma. Clustering of methylation data by tSNE/UMAP highlighted two highly distinct superclusters, with multiple subgroups within each. Supercluster_I was defined by radiation-induced secondary and/or hypermutant tumours, incorporating HGG-E (n = 44), cerebellar-enriched tumours (n = 38), and the paediatric RTK1 group (n = 295), further split into A, B and C subgroups. There were profound molecular differences between RTK1A and B/C subgroups, with 1A harbouring few CNAs and many more SNVs (SETD2, NF1), even in the absence of a hypermutator phenotype (also enriched compared to RTK1B/C), as well as a significantly longer overall survival. Distinct from these subgroups was Supercluster_II, which included pedHGG-RTK2A/B (n = 117), but also pedHGGA/B (n = 84) and pedHGG-MYCN (n = 122) subgroups, in addition to the predominantly H3-WT DMG-EGFR (n = 86). Although seemingly disparate, a common feature of these tumours was a highly infiltrative phenotype, either involving multiple cerebral lobes (gliomatosis cerebri) or thalami (bithalamic glioma). Analogous to pedHGG-RTK1, RTK2A harboured few CNAs and more CNVs (BCOR, PIK3CA), and a longer survival compared with 2B. Integrating subgroup-specific differential methylation and gene expression identified subgroup-specific epigenetic regulation of numerous developmentally-restricted transcription factors associated with their distinct neurodevelopmental origins; combining deconvolution approaches to bulk analyses with integrated scRNAseq allowed for identification of subgroup-specific immune cell annotation. Conclusion H3-WT-PDHGG segregate into two major classes with common clinical features, but each with multiple subgroups harbouring key molecular and phenotypic differences.

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)0.002
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: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.501
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.003
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.001
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 head0.014
GPT teacher head0.285
Teacher spread0.271 · 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