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Record W4212816860 · doi:10.1002/humu.24348

PhenomeCentral: 7 years of rare disease matchmaking

2022· article· en· W4212816860 on OpenAlexafffund
Matthew Osmond, Taila Hartley, Brittney Johnstone, Sasha Andjic, Marta Gîrdea, Meredith Gillespie, Orion J. Buske, Sergiu Dumitriu, Veronika Koltunova, Arun Ramani, Kym M. Boycott, Michael Brudno

Bibliographic record

VenueHuman Mutation · 2022
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsHospital for Sick ChildrenSunnybrook Health Science CentreUniversity of TorontoUniversity Health NetworkOntario Neurotrauma FoundationHealth Sciences CentreChildren's Hospital of Eastern OntarioUniversity of Ottawa
FundersCanadian Institutes of Health Research
KeywordsBiologyData sharingPhenotypeSet (abstract data type)Genotype-phenotype distinctionData setDiseaseComputational biologyGenomicsComputer scienceGenomeData scienceBioinformaticsGeneGeneticsArtificial intelligenceMedicine

Abstract

fetched live from OpenAlex

A major challenge in validating genetic causes for patients with rare diseases (RDs) is the difficulty in identifying other RD patients with overlapping phenotypes and variants in the same candidate gene. This process, known as matchmaking, requires robust data sharing solutions to be effective. In 2014 we launched PhenomeCentral, a RD data repository capable of collecting computer-readable genotypic and phenotypic data for the purposes of RD matchmaking. Over the past 7 years PhenomeCentral's features have been expanded and its data set has consistently grown. There are currently 1615 users registered on PhenomeCentral, which have contributed over 12,000 patient cases. Most of these cases contain detailed phenotypic terms, with a significant portion also providing genomic sequence data or other forms of clinical information. Matchmaking within PhenomeCentral, and with connections to other data repositories in the Matchmaker Exchange, have collectively resulted in over 60,000 matches, which have facilitated multiple gene discoveries. The collection of deep phenotypic and genotypic data has also positioned PhenomeCentral well to support next generation of matchmaking initiatives that utilize genome sequencing data, ensuring that PhenomeCentral will remain a useful tool in solving undiagnosed RD cases in the years to come.

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)0.096
metaresearch head score (Gemma)0.149
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 score0.096
Threshold uncertainty score0.507

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0960.149
Meta-epidemiology (narrow)0.0020.003
Meta-epidemiology (broad)0.0030.003
Bibliometrics0.0110.011
Science and technology studies0.0040.004
Scholarly communication0.0160.026
Open science0.0070.028
Research integrity0.0040.010
Insufficient payload (model declined to judge)0.0230.016

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 head0.010
GPT teacher head0.248
Teacher spread0.238 · 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

Citations18
Published2022
Admission routes2
Has abstractyes

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