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Record W7135057516 · doi:10.1093/neuped/wuaf001.122

EPEN-10. Open Sea DNA Hypomethylation/Island Hypermethylation ratio as a Predictor of Chromosomal Instability and Survival in PFA ependymoma

2025· article· en· W7135057516 on OpenAlexaff
Andrew M. Donson, Tzu Phang, Andrea M. Griesinger, Vladimir Amani, Enrique Grimaldo, Timothy Ritzmann, John-Paul Kilday, Kristian W Pajtler, David R. Ghasemi, Amy Smith, Nicholas K. Foreman, Vijay Ramaswamy

Bibliographic record

VenueNeuro-Oncology Pediatrics · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCancer Genomics and Diagnostics
Canadian institutionsHospital for Sick Children
Fundersnot available
KeywordsDNA methylationMethylationChromosome instabilityCorrelationPhenotypeCpG siteDNA

Abstract

fetched live from OpenAlex

Abstract Posterior fossa group A ependymoma (PFA) recur in more than 50% of cases. At recurrence a significant number of cases acquire high-risk chromosomal copy number variants (CNV), specifically gain of chromosome 1q (1q+) and/or loss of 6q (6q-). Analysis of DNA methylation profiles at presentation showed a correlation between open sea hypomethylation and island hypermethylation with acquisition of 1q+ and/or 6q- at recurrence. We hypothesize that hypomethylation of open sea DNA methylation sites results in chromosomal instability in PFA, and subsequent acquisition of 1q+ and/or 6q-. Identification of 1q+/6q- predisposed cases at presentation would provide a tool for stratification into a high-risk arm for upcoming clinical trials. Using a training set of 1q+ and/or 6q- predisposed (n = 24) and non-predisposed (“stable”, n = 29) we developed a DNA methylation-based predictive classifier, predominantly based on the ratio of island to open sea methylation sites, that correctly classified 96% (51/53) of training set cases. The performance of this classifier was tested in an independent multi-institutional dataset of approximately 500 PFA for which DNA methylation from presentation samples and survival data were available. Although this dataset did not contain information regarding the CNV status of recurrences thus preventing our ability to test prediction of high-risk CNV acquisition, we could infer acquisition of a high-risk CNV phenotype by outcome analysis. Multivariate analysis of outcome from presentation showed that samples classed as “predisposed” had a significantly shorter overall survival than those classed as “stable”. This result suggests the PFA high-risk DNA methylation classifier will be of utility in identification of those cases that are predisposed to high-risk CNV acquisition. We hope to definitively test the classifier in correlative studies of both the COG ependymoma clinical trial ACNS0831and the European BIOMECA study (SIOP EPN II trial related) for which recurrence CNV status is pending.

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.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.003
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.007

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0020.000

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 head0.012
GPT teacher head0.280
Teacher spread0.269 · 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

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2025
Admission routes1
Has abstractyes

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