Dutch patients, caregivers and healthcare professionals generate first nationwide research agenda for juvenile idiopathic arthritis

• DOI: 10.1186/s12969-021-00540-2
• Publication Date: 2021-04-07T12:03:19Z
• Authors: Anouk Verwoerd; Wineke Armbrust; Katherine Cowan; Lotte van den Berg; Joke de Boer; Sanne Bookelman; Marjan Britstra; Jeannette Cappon; Maria Certan; Christine Dedding; Karin van den Haspel; Petra Hissink Muller; Karin Jongsma; Otto Lelieveld; Jorg van Loosdregt; Wendy Olsder; Johanna Rocha; Ellen Schatorjé; Natasja Schouten; Joost F. Swart; Sebastiaan Vastert; Margot Walter; Casper G. Schoemaker
• Keywords: Adult; Adolescent; Health Personnel; 610; Diseases of the musculoskeletal system; Pediatrics; RJ1-570; Young Adult; 03 medical and health sciences; 0302 clinical medicine; Rheumatology; Research priority setting; Immunology and Allergy; Humans; Paediatrics - Radboud University Medical Center; Pediatrics, Perinatology, and Child Health; Radboudumc 5: Inflammatory diseases RIMLS: Radboud Institute for Molecular Life Sciences; Patient involvement; Child; Netherlands; Juvenile idiopathic arthritis; Middle Aged; Arthritis, Juvenile; 3. Good health; RC925-935; Caregivers; Child, Preschool; Self Report; James Lind Alliance; Research Article

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.