Addressing cohort reliance in rare diseases literature: a scoping review of myositis research
- Journal Title
- Rheumatology
- Publication Type
- Jan 16
- Abstract
- OBJECTIVES: This scoping review aimed to evaluate the extent and impact of duplication bias within contemporary idiopathic inflammatory myopathy (IIM) research. METHODS: A systematic scoping review was conducted to identify original research articles on IIM published between January 1, 2021, and May 18, 2023 using MEDLINE and EMBASE databases. Among 2,789 retrieved articles, 380 studies met inclusion criteria. Extracted data included IIM subtype, cohort type, sample size, and study design. RESULTS: Registry-based investigations constituted 10.0% of total publications, indicating significant potential for duplication bias. The over-representation of specific cohorts was evident, particularly from China, Japan and the United States, with 33.4% from Asia, 16.58% from Europe and 6.84% from North America. CONCLUSION: Current myositis research demonstrates substantial dependence on a restricted set of study populations, with limited geographic and demographic diversity. The findings underscore the critical need for broader representation and transparent reporting of cohort sources in myositis research. Promoting rigorous standards in cohort referencing and fostering diverse registries are critical steps toward reducing systemic research biases and enhancing equitable representation across patient populations in IIM.
- Publisher
- Oxford Academic
- Keywords
- Cochrane; Myositis; Prisma; case control; cohort; observational; scoping review; systematic searches
- Research Division(s)
- Inflammation
- PubMed ID
- 41546378
- Publisher's Version
- https://doi.org/10.1093/rheumatology/keag013
- Terms of Use/Rights Notice
- Refer to copyright notice on published article.
Creation Date: 2026-01-29 02:00:45
Last Modified: 2026-01-29 02:01:03