Frambu Resource Center for Rare Disorders, Sandbakkveien 18, Siggerud, 1404, Norway.
Norwegian National Advisory Unit on Learning and Mastery in Health, Oslo University Hospital, Postboks 4959 Nydalen, Oslo, 0424, Norway.
Orphanet J Rare Dis. 2024 Sep 6;19(1):328. doi: 10.1186/s13023-024-03332-5.
The ability to find, understand, appraise and utilise health information is crucial among individuals living with rare disorders. The aim of this study was to give a comprehensive overview of the literature on health literacy in adult persons with rare disorders.
We applied a scoping review methodology and performed a systematic search in 2021 in bibliographic databases. Searches were conducted in Medline (Ovid), Embase (Ovid), PsycInfo (Ovid), CINAHL (ebsco), and ERIC (Ovid). References were sorted and evaluated for inclusion using EndNote and Covidence. This review was guided by the question "What are the characteristics of research on health literacy in rare disorders?"
The database searches yielded 75 eligible reports. A total of 6223 individuals with rare disorders were represented alongside 1707 caregivers. The reports in this review have included study participants representing a total of 80 different rare disorders with unique ORPHA and ICD-10 codes. The results revealed that persons with rare disorders often exhibit gaps in health literacy through a lack of knowledge and access to information related to self-management, their own diagnosis and health, as well as daily coping and social rights. In addition, the importance of aid and information from healthcare personnel and the significance of getting social support from others in the same situation were accentuated.
This review emphasizes the importance of reinforcing health literacy among persons with rare disorders through peer support and education. This is the first review to give a comprehensive and state-of-the-art overview of literature investigating health literacy among persons with rare disorders and offers a basis for further research.
在患有罕见疾病的个体中，查找、理解、评估和利用健康信息的能力至关重要。本研究旨在全面概述成人罕见疾病患者健康素养的文献。
我们采用了范围综述方法，并于 2021 年在文献数据库中进行了系统搜索。在 Medline（Ovid）、Embase（Ovid）、PsycInfo（Ovid）、CINAHL（ebsco）和 ERIC（Ovid）中进行了搜索。使用 EndNote 和 Covidence 对参考文献进行排序和评估，以确定是否包含在内。本综述的问题是“罕见疾病健康素养研究的特点是什么？”
数据库搜索产生了 75 份合格报告。共有 6223 名罕见疾病患者和 1707 名照顾者被纳入研究。本综述中的报告包括了代表 80 种不同罕见疾病的研究参与者，他们具有独特的 ORPHA 和 ICD-10 编码。结果表明，罕见疾病患者在健康素养方面经常存在差距，他们缺乏与自我管理、自身诊断和健康以及日常应对和社会权利相关的知识和获取信息的途径。此外，强调了医疗保健人员提供援助和信息以及从同一情况下的其他人获得社会支持的重要性。
本综述强调了通过同伴支持和教育来增强罕见疾病患者健康素养的重要性。这是第一篇全面概述调查罕见疾病患者健康素养的文献综述，并为进一步研究提供了基础。
