Objective To evaluate research priority setting approaches in childhood chronic diseases and to describe the priorities of stakeholders including patients, caregivers/families and health professionals.
Design We conducted a systematic review of MEDLINE, Embase, PsycINFO and CINAHL from inception to 16 October 2016. Studies that elicited stakeholder priorities for paediatric chronic disease research were eligible for inclusion. Data on the prioritisation process were extracted using an appraisal checklist. Generated priorities were collated into common topic areas.
Results We identified 83 studies (n=15 722). Twenty (24%) studies involved parents/caregivers and four (5%) children. The top three health areas were cancer (11%), neurology (8%) and endocrine/metabolism (8%). Priority topic areas were treatment (78%), disease trajectory (48%), quality of life/psychosocial impact (48%), disease onset/prevention (43%), knowledge/self-management (33%), prevalence (30%), diagnostic methods (28%), access to healthcare (25%) and transition to adulthood (12%). The methods included workshops, Delphi techniques, surveys and focus groups/interviews. Specific methods for collecting and prioritising research topics were described in only 60% of studies. Most reviewed studies were conducted in high-income nations.
Conclusions Research priority setting activities in paediatric chronic disease cover many discipline areas and have elicited a broad range of topics. However, child/caregiver involvement is uncommon, and the methods often lack clarity. A systematic and explicit process that involves patients and families in partnership may help to inform a more patient and family-relevant research agenda in paediatric chronic disease.
- outcomes research
- patient perspective
- qualitative research
- stakeholder engagement
- chronic disease
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Contributors HLO conceptualised the study, carried out the data collection and analysis, coding of data, drafted the initial manuscript, drafted the manuscript and approved the final manuscript as submitted. AT and PL-V conceptualised and designed the study, contributed to data collection and analysis, reviewed and revised the manuscript and approved the final manuscript as submitted. AD, AJ, AM, RP, PR, SC, PHYC and JC contributed to study design, data analysis, reviewed and revised the manuscript and approved the final manuscript as submitted. MW contributed to study design, reviewed and revised the manuscript and approved the final manuscript as submitted. BK contributed to study design, data collection and analysis, reviewed and revised the manuscript and approved the final manuscript submitted. JCC conceptualised and designed the study, contributed to the analysis, reviewed and revised the manuscript and approved the final manuscript as submitted. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
Funding HLO received a scholarship from the Better Treatment for Kids Network funded by NSW Health. AT is supported by the National Health and Medical Research Council Fellowship (1106716). Professor Wake was supported by NHMRC Senior Research Fellowship and Cure Kids New Zealand. Research at the Murdoch Children’s Research Institute research is supported by the Victorian Government’s Operational Infrastructure Program.
Competing interests None declared.
Patient consent Not required.
Provenance and peer review Not commissioned; externally peer reviewed.