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Record W4409212271 · doi:10.1093/neuonc/noaf092

Salvage therapies for first relapse of SHH medulloblastoma in early childhood

2025· article· en· W4409212271 on OpenAlexaff
Craig Erker, Martin Mynarek, Marie Simbozel, Brandon T. Craig, Virginia L. Harrod, Andréa Maria Cappellano, Kenneth J. Cohen, Vicente Santa‐María López, Andrés Morales-La Madrid, Chantel Cacciotti, Lorena Baroni, Ralph Salloum, Ashley Margol, George Michaiel, Dolly Aguilera, Claire Mazewski, Cassie Kline, Jonathan L. Finlay, Mohamed S Abdelbaki, Jeffrey C. Murray, Kathleen Dorris, Bruce Crooks, Kevin Ginn, Nisreen Amayiri, Stephan Tippelt, Gudrun Fleischhack, Svenja Tonn, Nicolas U. Gerber, Álvaro Lassaletta, Jordan R. Hansford, Sara Khan, Stephen W. Gilheeney, Lindsey M. Hoffman, Michal Zápotocký, Valérie Larouche, Shafqat Shah, Vijay Ramaswamy, Amar Gajjar, Sébastien Perreault, Sabine Mueller, Juliette Hukin, Sylvia Cheng, Zhihong Wang, Kara Matheson, Simon Bailey, Éric Bouffet, Steven C Clifford, Giles Robinson, Christelle Dufour, Stefan Rutkowski, Lucie Lafay‐Cousin

Bibliographic record

VenueNeuro-Oncology · 2025
Typearticle
Languageen
FieldMedicine
TopicGlioma Diagnosis and Treatment
Canadian institutionsSickKids FoundationUniversité de MontréalHospital for Sick ChildrenBC Children's HospitalUniversité LavalUniversity of British ColumbiaLondon Health Sciences CentreUniversity of TorontoWestern UniversityNova Scotia Health AuthorityIzaak Walton Killam Health CentreAlberta Children's HospitalUniversity of CalgaryChildren's Hospital of Western OntarioCentre Hospitalier Universitaire Sainte-JustineDalhousie University
Fundersnot available
KeywordsMedicineMedulloblastomaHazard ratioVismodegibRadiation therapyOncologyProportional hazards modelInternal medicineChemotherapySalvage therapyCohortSurgeryPathologyConfidence interval

Abstract

fetched live from OpenAlex

BACKGROUND: Sonic hedgehog (SHH) medulloblastoma is the most common molecular group of infant and early childhood medulloblastoma (iMB) and has no standard of care at relapse. This work aimed to evaluate the post-relapse survival (PRS) and explore prognostic factors of patients with nodular desmoplastic (ND) and/or SHH iMB. METHODS: This international retrospective study included 147 subjects diagnosed with relapsed ND/SHH iMB between 1995 and 2017, <6 years old at original diagnosis, and treated without initial craniospinal irradiation (CSI). Univariable and multivariable Cox models with propensity score analyses were used to assess PRS for those in the curative intent cohort. RESULTS: The 3-year PRS was 61.6% (95% confidence interval [CI], 52.2-69.6). The median age at relapse was 3.4 years (interquartile range [IQR], 2.6-4.1). Those with local relapse (40.8%) more often received salvage treatment with surgery (P < .001), low-dose CSI (≤24 Gy; P < .001), or focal radiotherapy (P = .008). Patients not receiving CSI (40.5%) more often received salvage marrow-ablative chemotherapy (HDC + AuHCR [P < .001]). On multivariable analysis, CSI was associated with improved survival (hazard ratio [HR] 0.33 [95% CI, 0.13-0.86], P = .04). Salvage HDC + AuHCR, while clinically important, did not reach statistical significance (HR 0.24 [95% CI, 0.0054-1.025], P = .065). CONCLUSIONS: Survival of patients with relapsed SHH iMB is not satisfactory and relies on treatments associated with toxicities including CSI and/or HDC + AuHCR. Cure at initial diagnosis to avoid relapse is crucial. For patients with localized relapse undergoing resection, alternative salvage regimens that avoid high-dose CSI (>24 Gy) can be considered.

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 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)0.000
Version: codex-gemma-dda1882f352aValidation 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 score0.188
Threshold uncertainty score0.461

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
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 head0.012
GPT teacher head0.283
Teacher spread0.272 · 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 teacher head, 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

Citations3
Published2025
Admission routes1
Has abstractyes

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