This project has brought patients, caregivers and clinicians together in creating a research agenda for JIA using the JLA method. This is one of the first times a PSP has integrated the JLA approach with additional focus groups with children to ensure involvement of paediatric
Table 4
The final top 10 research priorities for juvenile idiopathic arthritis
1. Pain and fatigue are often present when the disease is in remission. How does this happen, what can one do about it, and can one predict which patients will suffer from them? |
2. What is the best treatment plan for each individual patient? (e.g. start a biological directly, which one, and what to do when the first one does not work, and how can medication best be tapered off?) |
3. What is the best treatment plan for uveitis in JIA, and are there factors that predict its effectiveness? |
4. Why are children with JIA fatigued more quickly, what can be done about it, and how can one best cope with the fatigue in daily life? |
5. How does JIA develop and which factors influence this? |
6. How can the course (flares, extensions, cure) of JIA be better explained and predicted? |
7. What is the influence of nutrition on JIA, and can a diet help? |
8. What are the short and long term side effects/consequences of the drugs taken for JIA? |
9. What is the influence of JIA on future opportunities regarding school results, work and relationships? |
10. What is the influence of sports and exercise on JIA and vice versa? |
patients of all age categories. We found that the number one research priority involved the cause and care of pain and fatigue when the disease was in remission. This is in line with a qualitative study by Bromberg et al. that finds self-reported pain and fatigue are highly common in children with JIA despite advances in treatment strategies(17). It is also highlighted to be an important research area by Palman et al(18), along with defining better predictors of remission states, which was also part of our Top 10. The fact that these studies all underline the same top research priority demonstrates the importance of future research focusing on this. Next to pain and fatigue, the aetiology of the disease remains an important topic that was featured high in our Top 10 and also in a recent priority setting exercise in the United States(19).
Moreover, our study exposed other important research areas such as personalised treatment strategies, JIA-associated uveitis, nutrition, long-term effects of drugs taken for JIA and sports/exercise that were not directly featured in other studies. This may be due to the wide scope we defined for this project, as well as the easily accessible nature of the JLA method that may have led to the collection of more widespread themes. The fact that the questions do not only cover the healthcare setting but also comprise more (psycho)social and educational issues suggests funding for these topics can also be sought from psychological, social and educational funding bodies.
One major strength of our study is the use of the systematic and transparent JLA method, which enhances the validity of our results. Furthermore, our efforts to include younger children is a strength. We conducted focus groups during the two phases of the process where information was gathered, such that they truly had their say in determining the most important topics. Importantly, question 9 of the final Top 10 (which was the top priority for younger children) made it into the list because a young adult patient advocated for it during the final workshop, using the argument that it was the children’s top priority. Two questions of the children’s Top 5 made it to the final Top 10 (see Table 3), which indicates the significant influence they had in the PSP.
A challenge in this study was the inclusiveness of people with all educational backgrounds and age groups throughout the survey phases. We started to use hardcopy questionnaires in these phases, which improved the inclusion of respondents with regard to education level. The input of patients and parents in the steering group was also very valuable in our search to reach all age groups. For example, they thought of using a cartoon on the flyers and in social media coverage to capture people’s attention. Furthermore, it is important to realise that participants of the final workshop consisted of a (predominantly) group of white, articulate, higher educated people, despite explicit efforts to include people with lower educational backgrounds and other ethnicities. This may have influenced which questions made it to the Top 10. This is a typical tendency in survey research and was also observed in other PSPs(20). Future PSPs may want to come up with and employ alternative strategies to ensure equal representation of all members of society. We also observed that more questions from the clinicians initial shortlist made it into the final Top 10 (see Tables 3 and 4), despite the fact that the nominal group technique was used during the priority setting workshop to ensure everyone had equal speaking time (even less outspoken people). This may be due to the fact that clinicians are more trained to give their opinion about these matters than young people and parents. Nevertheless, all members of the final workshop in the end agreed on the final Top 10.
Formally, this Top 10 was generated in the Netherlands, but we expect it also holds for other western countries. Now that the research agenda has been set, it is important that it is now indeed implemented with research funders and researchers. To achieve this, we attempted to inform the whole JIA research community in the Netherlands of this prioritisation exercise from the start, and some of the research groups made the commitment to truly incorporate the Top 10 in the long-term vision of their research lines. In addition, we have organised workshops with researchers, patients, parents and clinicians to turn the relatively broad questions into research proposals that can be presented to funding agencies. These workshops will take place in the fall of 2020. With these workshops, we hope to gather a group of people that is truly committed to finding answers to the research questions of the Top 10.