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Record W4407392464 · doi:10.1158/1078-0432.ccr-24-3278

Update on Cancer and Central Nervous System Tumor Surveillance in Pediatric <i>NF2</i> -, <i>SMARCB1</i> -, and <i>LZTR1</i> -Related Schwannomatosis

2025· article· en· W4407392464 on OpenAlexaff
Melissa R. Perrino, Marjolijn C.J. Jongmans, Gail E. Tomlinson, Mary‐Louise C. Greer, Sarah Scollon, Sarah G. Mitchell, Jordan R. Hansford, Kris Ann P. Schultz, Wendy Kohlmann, Jennifer M. Kalish, Suzanne P. MacFarland, Anirban Das, Kara N. Maxwell, Stefan M. Pfister, Rosanna Weksberg, Orli Michaeli, Uri Tabori, Gina M. Ney, Philip J. Lupo, Jack Brzezinski, Douglas R. Stewart, Emma R. Woodward, Christian P. Kratz

Bibliographic record

VenueClinical Cancer Research · 2025
Typearticle
Languageen
FieldMedicine
TopicNeurofibromatosis and Schwannoma Cases
Canadian institutionsHospital for Sick ChildrenUniversity of Toronto
FundersDivision of Cancer Epidemiology and Genetics, National Cancer InstituteManchester Biomedical Research CentreNational Cancer InstituteNational Institutes of HealthDeutsche KinderkrebsstiftungAmerican Lebanese Syrian Associated CharitiesNational Institute for Health and Care ResearchDepartment of Health and Social CareServierHospital Research FoundationAlexion Pharmaceuticals
KeywordsNeurofibromatosis type 2SMARCB1MedicineSchwannomaNeurofibromatosesMeningiomaAcoustic neuromaOncologyPathologyInternal medicineNeurofibromatosisSurgeryBiologyGene

Abstract

fetched live from OpenAlex

Schwannomatosis (SWN) is a distinct cancer predisposition syndrome caused by germline pathogenic variants in the genes NF2, SMARCB1, or LZTR1. There is a significant clinical overlap between these syndromes with the hallmark of increased risk for cranial, spinal, and peripheral schwannomas. Neurofibromatosis type 2 was recently renamed as NF2-related SWN and is the most common SWN syndrome, with increased risk for bilateral vestibular schwannomas, intradermal schwannomas, meningiomas, and less commonly, ependymoma. SMARCB1-related SWN is a familial SWN syndrome associated with peripheral and spinal schwannomas and an increased risk for meningiomas and malignant peripheral nerve sheath tumors, even in the absence of radiation. These individuals do not develop bilateral vestibular schwannomas. Finally, patients with LZTR1-related SWN typically present with peripheral schwannomas, and unilateral vestibular schwannomas have been reported. The following perspective is intended to highlight the clinical presentation and international tumor surveillance recommendations across these SWN syndromes.

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)0.002
metaresearch head score (Gemma)0.007
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: none
GenreCandidate signal: Empirical · Consensus signal: none
Teacher disagreement score0.006
Threshold uncertainty score0.013

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.007
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0040.004
Science and technology studies0.0000.000
Scholarly communication0.0010.002
Open science0.0010.001
Research integrity0.0020.003
Insufficient payload (model declined to judge)0.0030.002

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 head0.058
GPT teacher head0.413
Teacher spread0.355 · 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

Citations8
Published2025
Admission routes1
Has abstractyes

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