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Record W4385458457 · doi:10.1080/21678707.2023.2241347

Real world data for rare diseases research: The beginner’s guide to registries

2023· article· en· W4385458457 on OpenAlexaff
Federica Pisa, Ariel E. Arias, Emily W. Bratton, Maribel Salas, Janet Sultana

Bibliographic record

VenueExpert Opinion on Orphan Drugs · 2023
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Rare Diseases
Canadian institutionsUniversité de Montréal
Fundersnot available
KeywordsMedicineDiseaseMedical recordRare diseaseSummary of Product CharacteristicsDisease registryMEDLINEProduct (mathematics)Family medicineMedical emergencyData scienceDrugPathologyComputer sciencePharmacology

Abstract

fetched live from OpenAlex

Introduction Rare disease research has specific challenges that can be addressed using registries.Areas covered There are at least three different types of registries: patient registries, disease registries, and product registries. Patient registries recruit rare disease patients, potentially including several rare diseases within a registry, while disease registries can be considered a subset of patient registries which focus on specific diseases. Product registries focus on specific drugs. These registries may be used to conduct research that is specifically requested by a regulatory authority, they may be developed by a drug company to monitor the use of a particular drug or may be developed for public health monitoring purposes.Expert Opinion Compared to other sources of real-world data (RWD), such as electronic medical records (EMRs) and claims data, registries are more likely to have a correct diagnosis and more specific information about RDs. However, registries also have their challenges. Competition between registries may lead to missing or incomplete data. Registries could also have limited information on drug and medical history, which are better captured in EMRs or claims. Nevertheless, registries remain an important source of RWD in the rare disease space and are increasingly being leveraged to comply with regulatory requirements.

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)0.000
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.262
Threshold uncertainty score0.510

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.001
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 head0.111
GPT teacher head0.423
Teacher spread0.313 · 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 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

Citations12
Published2023
Admission routes1
Has abstractyes

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