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Record W4410878401 · doi:10.1016/j.xhgg.2025.100462

Breaking barriers in rare disease research: The RARE-X Open Science Data Challenge as a model for collaborative innovation and community partnership

2025· article· en· W4410878401 on OpenAlexaff
Karmen M Trzupek, Ravi Bhargava, Fanny Sie, Vanessa Vogel‐Farley, Verena Chung, María Díaz, Charlene Son-Rigby, Joseph Geraci, Jacob Albrecht

Bibliographic record

VenueHuman Genetics and Genomics Advances · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsDuPont (Canada)Roche (Canada)
FundersHorizon TherapeuticsF. Hoffmann-La RocheRoche
KeywordsGeneral partnershipRare diseaseOpen scienceEngineering ethicsDiseaseKnowledge managementPolitical scienceSociologyMedicinePublic relationsEngineeringComputer sciencePhysicsPathology

Abstract

fetched live from OpenAlex

Trzupek et al. describe a rare disease Open Science Data Challenge, using data collected systematically on RARE-X across 27 neurodevelopmental disorders. Clinical diagnoses, symptoms, genetic data, and PROs were included. Researchers and statisticians generated solutions that identified previously underappreciated symptoms and used machine learning to test predictive models for diagnosis.

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)0.367
metaresearch head score (Gemma)0.509
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesMetaresearch, Open science
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: none
Teacher disagreement score0.993
Threshold uncertainty score0.780

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.3670.509
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0030.003
Bibliometrics0.0050.005
Science and technology studies0.0110.021
Scholarly communication0.0300.046
Open science0.0070.043
Research integrity0.0170.032
Insufficient payload (model declined to judge)0.0080.002

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 head0.166
GPT teacher head0.434
Teacher spread0.268 · 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.

Study designNot applicable
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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