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Record W4402052589 · doi:10.51731/cjht.2024.961

An Inventory of Rare Disease Registries in the Canadian Landscape

2024· article· en· W4402052589 on OpenAlexaboutno aff
CADTH

Bibliographic record

VenueCanadian Journal of Health Technologies · 2024
Typearticle
Languageen
FieldEnvironmental Science
TopicEnvironmental DNA in Biodiversity Studies
Canadian institutionsnot available
Fundersnot available
KeywordsGeography

Abstract

fetched live from OpenAlex

What Was the Question? There is a lack of centralized information about rare disease registries (RDRs) in Canada. To address this, we created an inventory of RDRs in Canada and international RDRs that include patients living in Canada. What Did We Do? Information about RDRs was identified through a search of published literature, grey literature, consultations with members of the rare disease community, and a survey with the registry holders. What Did We Find? We identified 148 RDRs, of which 66 are RDRs in Canada and 82 international RDRs. In total, 21% of RDRs in Canada and 11% of international RDRs capture rare cancer(s). About a half of RDRs in Canada (53%) and international RDRs (46%) collaborate with other national or international registries or networks. Most RDRs in Canada (73%) and international RDRs (86%) identify patients for RDR enrollment through health care provider referrals. Electronic medical records (68%), clinician-reported data (68%), and medical chart abstraction (60%) are the most common sources of data for RDRs in Canada compared to international RDRs, for which patient (79%) and caregiver (61%) surveys are most common. Most RDRs in Canada collect clinical data (95%), laboratory and diagnostic data (85%), health outcomes data (83%), and treatment data (78%), and less commonly patient-generated data (55%) and caregiver data (15%). What Does This Mean? The information in the inventory will help guide future initiatives for improving the RDR landscape in Canada. Most RDRs in Canada source data from electronic health records, clinician-reported data, and medical charts. Fewer RDRs in Canada implement patient and caregiver surveys. RDRs in Canada include information relevant to decision-makers as most collect clinical data, health outcomes data, and treatment data, and about half collect health resource utilization data. This inventory will support future initiatives to assess the suitability of RDRs for generating decision-grade real-world evidence.

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)0.001
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 score0.490
Threshold uncertainty score0.687

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0010.000
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 head0.022
GPT teacher head0.241
Teacher spread0.219 · 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
Published2024
Admission routes1
Has abstractyes

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