A scoping review of health literacy in rare disorders: key issues and research directions

被引：1

作者：

Stenberg, Una ^{[1
,2
]}

Westfal, Lydia ^{[1
]}

Dybesland Rosenberger, Andreas ^{[3
]}

Orstavik, Kristin ^{[4
]}

Flink, Maria ^{[5
]}

Holmen, Heidi ^{[6
]}

Systad, Silje ^{[7
]}

Westermann, Karl Fredrik ^{[2
]}

Velvin, Gry ^{[8
]}

机构：

[1] Frambu Resource Ctr Rare Disorders, Sandbakkveien 18, N-144 Siggerud, Norway

[2] Oslo Univ Hosp, Norwegian Natl Advisory Unit Learning & Mastery H, Postboks 4959 Nydalen, N-0424 Oslo, Norway

[3] Univ Hosp North Norway, Natl Neuromuscular Ctr Norway, N-9019 Tromso, Norway

[4] Oslo Univ Hosp, Dept Neurol, Unit Congenital & Hereditary Neuromuscular Disorde, Sect Rare Neuromuscular Disorders, 4950 Nydalen, N-0424 Oslo, Norway

[5] Karolinska Inst, Dept Neurobiol Care Sci & Soc, Solnavagen 1, S-17177 Solna, Sweden

[6] Oslo Metropolitan Univ, Postbox 4,St Olavs Pl, N-0130 Oslo, Norway

[7] Oslo Univ Hosp, Natl Ctr Rare Epilepsy Related Disorders, Dept Rare Disorders, Div Paediat & Adolescent Med, 4950 Nydalen, N-0424 Oslo, Norway

[8] Sunnaas Rehabil Hosp, TRS Resource Ctr Rare Dis, Bjornemyrveien 11, N-1453 Bjornemyr, Norway

来源：

ORPHANET JOURNAL OF RARE DISEASES | 2024年 / 19卷 / 01期

关键词：

Health literacy; Coping; Rare disorders; Rare disease; Rare genetic disorder; Rare developmental defect; Integrative model of health literacy; Scoping review; QUALITY-OF-LIFE; SPINA-BIFIDA; EXPERIENCES; DISEASE; ADULTS; INFORMATION; KNOWLEDGE; SUPPORT; INDIVIDUALS; HEMOPHILIA;

D O I：

10.1186/s13023-024-03332-5

中图分类号：

Q3 [遗传学];

学科分类号：

071007 ; 090102 ;

摘要：

BackgroundThe 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.MethodsWe 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?"ResultsThe 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.ConclusionThis 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.

引用

页数：11

共 96 条

[1] Exploring the experiences of having Guillain-Barre Syndrome: A qualitative interview study [J].