Barriers and facilitators to designing, maintaining, and utilizing rare disease patient registries: a scoping review protocol.
Journal
JBI evidence synthesis
ISSN: 2689-8381
Titre abrégé: JBI Evid Synth
Pays: United States
ID NLM: 101764819
Informations de publication
Date de publication:
21 Oct 2024
21 Oct 2024
Historique:
medline:
21
10
2024
pubmed:
21
10
2024
entrez:
21
10
2024
Statut:
aheadofprint
Résumé
The objectives of this review are to identify barriers/facilitators to designing, maintaining, and utilizing rare disease patient registries (RDPRs); determine whether and how these differ among patient partners, other knowledge users (KUs), and researchers; and chart definitions of rare diseases and RDPRs. RDPRs are vital to improving the understanding of the natural histories and predictors of outcomes for rare diseases, assessing interventions, and identifying potential participants for clinical trials. Currently, however, the functionality of RDPRs is not fully optimized. To improve the quality and functionality of RDPRs, it is important to understand the barriers and/or facilitators involved in their design, maintenance, and utilization; how these might differ among patient partners, other KUs, and researchers; and to delineate the range of definitions for rare diseases and RDPRs. Evidence of any study design or format (including empirical studies, books, manuals, commentaries, editorials, guidance documents, conference abstracts, review documents, and gray literature) referencing barriers/facilitators for designing, maintaining, or utilizing RDPRs will be considered for inclusion. The review will follow the JBI methodology for scoping reviews. We will search health science databases, including the Cochrane Library, Embase, MEDLINE, the JBI EBP Database, and PsycINFO, from inception onwards, as well as gray literature using the Canadian Agency for Drugs and Technologies in Health (CADTH) Grey Matters guidance. Two independent reviewers will screen titles and abstracts and full-text documents, as well as abstract data. Disagreements will be resolved through discussion or with a third reviewer. Evidence will be synthesized descriptively and reported using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews (PRIMSA-ScR). Open Science Framework https://osf.io/mvf9r.
Identifiants
pubmed: 39428992
doi: 10.11124/JBIES-24-00091
pii: 02174543-990000000-00373
doi:
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Informations de copyright
Copyright © 2024 JBI.
Déclaration de conflit d'intérêts
ACT is a member of the JBI Evidence Synthesis editorial board but was not involved in the editorial decision-making for this paper. The other authors declare no conflict of interest.
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