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Research priority setting in childhood chronic disease: a systematic review
  1. Harrison Lindsay Odgers1,2,
  2. Allison Tong1,2,
  3. Pamela Lopez-Vargas2,3,
  4. Andrew Davidson4,5,6,
  5. Adam Jaffe7,8,
  6. Anne McKenzie9,
  7. Ross Pinkerton10,
  8. Melissa Wake4,6,11,
  9. Peter Richmond12,13,
  10. Sally Crowe14,
  11. Patrina Ha Yuen Caldwell2,15,16,
  12. Sophie Hill17,
  13. Jennifer Couper18,19,
  14. Suzy Haddad20,
  15. Behrouz Kassai2,21,
  16. Jonathan C Craig1,2
  1. 1 Sydney School of Public Health, University of Sydney, Sydney, New South Wales, Australia
  2. 2 Centre for Kidney Research, The Children’s Hospital at Westmead, Westmead, New South Wales, Australia
  3. 3 Kid’s Research Institute, The Children’s Hospital at Westmead, Sydney, New South Wales, Australia
  4. 4 Department of Paediatrics, University of Melbourne, Melbourne, Victoria, France
  5. 5 Department of Anaesthesiology, Royal Children’s Hospital, Melbourne, Victoria, Australia
  6. 6 Murdoch Children’s Research Institute, Parkville, Victoria, Australia
  7. 7 Department of Respiratory Medicine, The Sydney Children’s Hospital Network, Sydney, New South Wales, Australia
  8. 8 Discipline of Paediatrics, The University of New South Wales, Sydney, New South Wales, Australia
  9. 9 Western Australian Health Translation Network, The University of Western Australia, Perth, Western Australia, Australia
  10. 10 Department of Oncology, Lady Cilento Children’s Hospital, South Brisbane, Queensland, Australia
  11. 11 Department of Paediatrics, The Liggins Institute, The University of Auckland, Auckland, New Zealand
  12. 12 Division of Paediatrics, School of Medicine, The University of Western Australia, Perth, Western Australia, Australia
  13. 13 Departments of General Paediatrics and Immunology, Princess Margaret Hospital for Children, Perth, Western Australia, Australia
  14. 14 Crowe Associated Ltd, Oxon, UK
  15. 15 Discipline of Paediatrics and Child Health, University of Sydney, Sydney, New South Wales, Australia
  16. 16 Department of Nephrology, The Children’s Hospital at Westmead, Sydney, New South Wales, Australia
  17. 17 Center for Health Communication and Participation, La Trobe University, Melbourne, Victoria, Australia
  18. 18 Department of Endocrinology and Diabetes, Women’s and Children’s Hospital, Adelaide, South Australia, Australia
  19. 19 Robinson Research Institute, University of Adelaide, Adelaide, South Australia, Australia
  20. 20 Patient and Carer Representative, Sydney, New South Wales, Australia
  21. 21 Centre d’Investigation Clinique de Lyon, Lyon, France
  1. Correspondence to Dr Harrison Lindsay Odgers, Centre for Kidney Research, The Children’s Hospital at Westmead, Sydney, NSW 2145, Australia; hodg7590{at}uni.sydney.edu.au

Abstract

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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Footnotes

  • 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.

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