Anouk Verwoerd1,2, Wineke Armbrust3,4, Katherine Cowan5, Lotte van den Berg6, Joke de Boer7, Sanne Bookelman6, Marjan Britstra6, Jeannette Cappon8,9, Maria Certan2,10, Christine Dedding11, Karin van den Haspel6, Petra Hissink Muller4,12, Karin Jongsma13, Otto Lelieveld9,14, Jorg van Loosdregt1,2, Wendy Olsder15, Johanna Rocha15, Ellen Schatorjé4,16,17, Natasja Schouten6, Joost F Swart2,4, Sebastiaan Vastert1,2,4, Margot Walter9,18, Casper G Schoemaker19,20,21. 1. Centre for Translational Immunology, University Medical Centre Utrecht, Heidelberglaan 100, 3584, CX, Utrecht, The Netherlands. 2. Department of Paediatric Immunology and Rheumatology, Wilhelmina Children's Hospital, Lundlaan 6, 3584, EA, Utrecht, The Netherlands. 3. University of Groningen, University Medical Centre Groningen, Department of Paediatric Rheumatology and Immunology, Beatrix Children's Hospital, Hanzeplein 1, 9713, GZ, Groningen, The Netherlands. 4. Dutch Society for Paediatric Rheumatology (NVKR), Mercatorlaan 1200, 3528, BL, Utrecht, The Netherlands. 5. James Lind Alliance, National Institute for Health Research Evaluation, Trials and Studies Coordinating Centre (NETSCC), based at the University of Southampton, Alpha House, Enterprise Road, Southampton, SO16 7NS, UK. 6. Dutch JIA Patient and Parent Organisation (JVN), member of ENCA, Pius X-straat 49, 5121, EP, Rijen, The Netherlands. 7. Department of Ophthalmology, University Medical Centre Utrecht, Heidelberglaan 100, 3584, CX, Utrecht, The Netherlands. 8. Reade, Centre for Rehabilitation and Rheumatology, Dr. Jan van Breemenstraat 2, 1056, AB, Amsterdam, The Netherlands. 9. Dutch Health Professionals in Paediatric Rheumatology (NHPKR), Amsterdam, The Netherlands. 10. Department of Science, University College Roosevelt, Lange Noordstraat 1, 4331, CB, Middelburg, The Netherlands. 11. Department of Medical Humanities, Amsterdam University Medical Centre, De Boelelaan 1117, 1081, HV, Amsterdam, The Netherlands. 12. Department of Paediatric Immunology and Rheumatology, Willem-Alexander Children's Hospital, Albinusdreef 2, 2333, ZA, Leiden, The Netherlands. 13. Julius Centre for Health Sciences and Primary Care, University Medical Centre Utrecht, Heidelberglaan 100, 3584, CX, Utrecht, The Netherlands. 14. University of Groningen, University Medical Centre Groningen, Department of Rehabilitation Medicine, Hanzeplein 1, 9713, GZ, Groningen, The Netherlands. 15. Youth-R-Well.com, Patient Organisation for Young Patients, member of EULAR PARE, Eikstraat 3, 3434, TD, Nieuwegein, The Netherlands. 16. Department of Paediatric Immunology and Rheumatology, Amalia Children's Hospital, Geert Grooteplein Zuid 10, 6525, GA, Nijmegen, The Netherlands. 17. Department of Paediatric Rheumatology, St. Maartenskliniek, Dokter Kopstraat 1, 5835, DV, Beugen, The Netherlands. 18. Department of Rheumatology, Erasmus University Medical Centre, Doctor Molewaterplein 40, 3015, GD, Rotterdam, The Netherlands. 19. Department of Paediatric Immunology and Rheumatology, Wilhelmina Children's Hospital, Lundlaan 6, 3584, EA, Utrecht, The Netherlands. c.g.schoemaker-4@umcutrecht.nl. 20. Dutch JIA Patient and Parent Organisation (JVN), member of ENCA, Pius X-straat 49, 5121, EP, Rijen, The Netherlands. c.g.schoemaker-4@umcutrecht.nl. 21. Faculty of Medicine, Utrecht University, Universiteitsweg 98, 3584, CG, Utrecht, The Netherlands. c.g.schoemaker-4@umcutrecht.nl.
Abstract
BACKGROUND: Involving the end-users of scientific research (patients, carers and clinicians) in setting research priorities is important to formulate research questions that truly make a difference and are in tune with the needs of patients. We therefore aimed to generate a national research agenda for Juvenile Idiopathic Arthritis (JIA) together with patients, their caregivers and healthcare professionals through conducting a nationwide survey among these stakeholders. METHODS: The James Lind Alliance method was used, tailored with additional focus groups held to involve younger patients. First, research questions were gathered through an online and hardcopy survey. The received questions that were in scope were summarised and a literature search was performed to verify that questions were unanswered. Questions were ranked in the interim survey, and the final top 10 was chosen during a prioritisation workshop. RESULTS: Two hundred and seventy-eight respondents submitted 604 questions, of which 519 were in scope. Of these 604 questions, 81 were generated in the focus groups with younger children. The questions were summarised into 53 summary questions. An evidence checking process verified that all questions were unanswered. A total of 303 respondents prioritised the questions in the interim survey. Focus groups with children generated a top 5 of their most important questions. Combining this top 5 with the top 10s of patients, carers, and clinicians led to a top 21. Out of these, the top 10 research priorities were chosen during a final workshop. Research into pain and fatigue, personalised treatment strategies and aetiology were ranked high in the Top 10. CONCLUSIONS: Through this study, the top 10 research priorities for JIA of patients, their caregivers and clinicians were identified to inform researchers and research funders of the research topics that matter most to them. The top priority involves the treatment and mechanisms behind persisting pain and fatigue when the disease is in remission.
BACKGROUND: Involving the end-users of scientific research (patients, carers and clinicians) in setting research priorities is important to formulate research questions that truly make a difference and are in tune with the needs of patients. We therefore aimed to generate a national research agenda for Juvenile Idiopathic Arthritis (JIA) together with patients, their caregivers and healthcare professionals through conducting a nationwide survey among these stakeholders. METHODS: The James Lind Alliance method was used, tailored with additional focus groups held to involve younger patients. First, research questions were gathered through an online and hardcopy survey. The received questions that were in scope were summarised and a literature search was performed to verify that questions were unanswered. Questions were ranked in the interim survey, and the final top 10 was chosen during a prioritisation workshop. RESULTS: Two hundred and seventy-eight respondents submitted 604 questions, of which 519 were in scope. Of these 604 questions, 81 were generated in the focus groups with younger children. The questions were summarised into 53 summary questions. An evidence checking process verified that all questions were unanswered. A total of 303 respondents prioritised the questions in the interim survey. Focus groups with children generated a top 5 of their most important questions. Combining this top 5 with the top 10s of patients, carers, and clinicians led to a top 21. Out of these, the top 10 research priorities were chosen during a final workshop. Research into pain and fatigue, personalised treatment strategies and aetiology were ranked high in the Top 10. CONCLUSIONS: Through this study, the top 10 research priorities for JIA of patients, their caregivers and clinicians were identified to inform researchers and research funders of the research topics that matter most to them. The top priority involves the treatment and mechanisms behind persisting pain and fatigue when the disease is in remission.
Entities:
Keywords:
James Lind Alliance; Juvenile idiopathic arthritis; Patient involvement; Research priority setting
Authors: Harrison Lindsay Odgers; Allison Tong; Pamela Lopez-Vargas; Andrew Davidson; Adam Jaffe; Anne McKenzie; Ross Pinkerton; Melissa Wake; Peter Richmond; Sally Crowe; Patrina Ha Yuen Caldwell; Sophie Hill; Jennifer Couper; Suzy Haddad; Behrouz Kassai; Jonathan C Craig Journal: Arch Dis Child Date: 2018-04-11 Impact factor: 3.791
Authors: Malcolm R Macleod; Susan Michie; Ian Roberts; Ulrich Dirnagl; Iain Chalmers; John P A Ioannidis; Rustam Al-Shahi Salman; An-Wen Chan; Paul Glasziou Journal: Lancet Date: 2014-01-08 Impact factor: 79.321
Authors: Casper G Schoemaker; Wineke Armbrust; Joost F Swart; Sebastiaan J Vastert; Jorg van Loosdregt; Anouk Verwoerd; Caroline Whiting; Katherine Cowan; Wendy Olsder; Els Versluis; Rens van Vliet; Marlous J Fernhout; Sanne L Bookelman; Jeannette Cappon; J Merlijn van den Berg; Ellen Schatorjé; Petra C E Hissink Muller; Sylvia Kamphuis; Joke de Boer; Otto T H M Lelieveld; Janjaap van der Net; Karin R Jongsma; Annemiek van Rensen; Christine Dedding; Nico M Wulffraat Journal: Pediatr Rheumatol Online J Date: 2018-09-15 Impact factor: 3.054