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Record W4399527680 · doi:10.1158/1078-0432.ccr-24-0237

Neuroblastoma Predisposition and Surveillance—An Update from the 2023 AACR Childhood Cancer Predisposition Workshop

2024· review· en· W4399527680 on OpenAlexaff
Junne Kamihara, Lisa Diller, William D. Foulkes, Orli Michaeli, Yoshiko Nakano, Kristian W. Pajtler, Melissa R. Perrino, Sarah Scollon, Douglas R. Stewart, Stephan D. Voss, Rosanna Weksberg, Jordan R. Hansford, Garrett M. Brodeur

Bibliographic record

VenueClinical Cancer Research · 2024
Typereview
Languageen
FieldMedicine
TopicNeuroblastoma Research and Treatments
Canadian institutionsSickKids FoundationHospital for Sick ChildrenMcGill UniversityUniversity of TorontoJewish General Hospital
FundersDivision of Cancer Epidemiology and Genetics, National Cancer InstituteBundesministerium für Bildung und ForschungServierModernaNational Cancer InstituteDeutsche KinderkrebsstiftungIncyteHospital Research FoundationAlexion PharmaceuticalsAstraZenecaSt. Baldrick's FoundationSanofi
KeywordsGenetic predispositionMedicineNeuroblastomaGermlineCancerGenetic testingCostello syndromeLi–Fraumeni syndromeDiseaseHypoventilationPediatric cancerGeneticsGermline mutationInternal medicineGeneMutationBiology

Abstract

fetched live from OpenAlex

Genetic predisposition to neuroblastoma (NB) is relatively rare. Only 1% to 2% of patients have a family history of NB, 3% to 4% of cases present with bilateral or multifocal primary tumors, and occasional patients have syndromes that are associated with increased NB risk. Previously, a germline pathogenic variant (GPV) in PHOX2B was associated with Hirschsprung disease and congenital central hypoventilation syndrome. Recently, certain GPVs were shown to be responsible for congenital central hypoventilation syndrome and NB predisposition. Also, several groups determined that activating GPVs in ALK accounted for a substantial number of familial NB. Finally, there are additional genes and cancer predisposition syndromes in which NB occurs with greater frequency or that have been associated with NB based on genome-wide association studies. We review the evidence for all these genes and whether there is sufficient evidence to warrant surveillance. We review recommended surveillance for hereditary patients with NB, including minor updates to surveillance recommendations that were published previously in 2017.

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)0.002
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow), Research integrity
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.949
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0020.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0020.001
Bibliometrics0.0000.001
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0010.001
Research integrity0.0010.005
Insufficient payload (model declined to judge)0.0010.001

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 head0.226
GPT teacher head0.557
Teacher spread0.330 · 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.

Study designNot applicable
Domainnot available
GenreReview

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

Citations23
Published2024
Admission routes1
Has abstractyes

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