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Record W7135089200 · doi:10.1093/neuped/wuaf001.247

QOL-01. Curing Sonic Hedgehog (SHH) Medulloblastoma without Irradiation: Investigating Late Effects in Survivorship

2025· article· en· W7135089200 on OpenAlex
Sameera Ramjan, Joseph Stanek, Girish Dhall, Brenna C. McDonald, Parth Patel, Megan Blue, Jonathan L. Finlay, Stephen Sands

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.

aboutThe title or abstract carries a Canadian signal from the geographic lexicon.
no affNo Canadian affiliation: this work is invisible to an affiliation-only frame.
No Canadian affiliation. An affiliation-only frame, the usual design, would never have seen this work. It is one of the works that make the case for inverting the frame.

Bibliographic record

VenueNeuro-Oncology Pediatrics · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicHedgehog Signaling Pathway Studies
Canadian institutionsnot available
Fundersnot available
KeywordsMedulloblastomaNeurocognitiveSonic hedgehogSurvivorship curveQuality of life (healthcare)Young adultCognitionPerformance status

Abstract

fetched live from OpenAlex

Abstract Introduction Cranial irradiation, a mainstay in medulloblastoma treatment, can be associated with neurocognitive late effects: lowered attention, working memory, processing speed, executive functioning, and learning, with young age at diagnosis a risk factor. By eliminating cranial irradiation for young, low-risk patients, will neurocognitive and quality of life morbidities decrease? Methods Patients with newly diagnosed SHH medulloblastoma enrolled on the Head Start 4 trial between 2016-2021 at 25 participating institutions in the US, Canada and New Zealand underwent baseline assessment prior to consolidation and autologous hematopoietic progenitor cell rescue and again at two-year-follow-up. Serial standardized test scores were compared using a linear mixed model with restricted maximum likelihood estimates and a random intercept for each patient. Results Thirty-two patients (median age 2.3 years, 68.6% male) completed baseline evaluation, and 21 (91% compliance rate) survivors on study were reassessed at 2-year follow-up. IQ and working memory remained stable over time (WPPSI-IV/WISC-V p = 0.71; p = 0.99, respectively). Similarly, overall quality of life (PedsQL p = 0.31), overall executive functioning (BRIEF GEC p = 0.08), overall adaptive functioning (ABAS-III GEC p = 0.10), adaptive skills (BASC-3 p = 0.65) and internalizing problems (BASC-3 p = 0.36) did not significantly change over time. In contrast, participants displayed increased attention difficulties (BASC-3 p = 0.02), decreased social quality of life (PedsQL p = 0.04), and lower practical skills at home (ABAS-3 p = 0.04). Discussion These novel results provide encouraging follow-up data for young patients treated for SHH Medulloblastoma without cranial irradiation. At the same time, areas such as attention, socialization, and practical skills at home appear to be impacted. These findings need to be further substantiated with continued monitoring through biannual assessments and additional trials, such as an upcoming phase III international randomized study. Specifically, two effective irradiation-sparing treatment regimens will be compared - Head Start 4 and HIT-SKK - across Europe and North America, to harmonize diagnostic and therapeutic standards for medulloblastoma.

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)0.003
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.075
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.003
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 science0.0000.000
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 head0.013
GPT teacher head0.280
Teacher spread0.267 · 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