Scoping review of patient- and family-oriented outcomes and measures for chronic pediatric disease

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Title: Scoping review of patient- and family-oriented outcomes and measures for chronic pediatric disease
Authors: Khangura, Sara D
Karaceper, Maria D
Trakadis, Yannis
Mitchell, John J
Chakraborty, Pranesh
Tingley, Kylie
Coyle, Doug
Grosse, Scott D
Kronick, Jonathan B
Laberge, Anne-Marie
Little, Julian
Prasad, Chitra
Sikora, Lindsey
Siriwardena, Komudi
Sparkes, Rebecca
Speechley, Kathy N
Stockler, Sylvia
Wilson, Brenda J
Wilson, Kumanan
Zayed, Reem
Potter, Beth K
Date: 2015-02-13
Abstract: Abstract Background Improvements in health care for children with chronic diseases must be informed by research that emphasizes outcomes of importance to patients and families. To support a program of research in the field of rare inborn errors of metabolism (IEM), we conducted a broad scoping review of primary studies that: (i) focused on chronic pediatric diseases similar to IEM in etiology or manifestations and in complexity of management; (ii) reported patient- and/or family-oriented outcomes; and (iii) measured these outcomes using self-administered tools. Methods We developed a comprehensive review protocol and implemented an electronic search strategy to identify relevant citations in Medline, EMBASE, DARE and Cochrane. Two reviewers applied pre-specified criteria to titles/abstracts using a liberal accelerated approach. Articles eligible for full-text review were screened by two independent reviewers with discrepancies resolved by consensus. One researcher abstracted data on study characteristics, patient- and family-oriented outcomes, and self-administered measures. Data were validated by a second researcher. Results 4,118 citations were screened with 304 articles included. Across all included reports, the most-represented diseases were diabetes (35%), cerebral palsy (23%) and epilepsy (18%). We identified 43 unique patient- and family-oriented outcomes from among five emergent domains, with mental health outcomes appearing most frequently. The studies reported the use of 405 independent self-administered measures of these outcomes. Conclusions Patient- and family-oriented research investigating chronic pediatric diseases emphasizes mental health and appears to be relatively well-developed in the diabetes literature. Future research can build on this foundation while identifying additional outcomes that are priorities for patients and families.
URL: http://dx.doi.org/10.1186/s12887-015-0323-x
http://hdl.handle.net/10393/33656
CollectionLibre accès - Publications // Open Access - Publications
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