What Are the Consequences of Not Responding to Research Priority Setting Exercises?

Field: Health Professions (Health Sciences)

The James Lind Alliance (JLA) was established in the United Kingdom in 2004 to enable the end users of health research, patients, carers and clinicians, to propose and agree on the most important topics in need of research.1 Since its establishment, there have been over 100 completed priority setting partnerships (PSPs), and the reach has extended internationally, where the JLA methodology is also being followed (see https://www.jla.nihr.ac.uk/priority-setting-partnerships/). Recently announced is a review of PSPs undertaken during 2016-2020, mapping out the information gained, looking for common themes and outputs (https://www.phc.ox.ac.uk/research/interdisciplinary-research-in-health-sciences/prioritising-health-research). Ahead of that review, having been involved in 2 of these exercises, we wanted to offer up some of our own reflections. To remind readers less familiar with the process, what it means to actually undertake a PSP is you: follow the JLA guidebook, which means keeping to its guideline, reviewing evidence, and engaging a JLA facilitator to support the process; establish a PSP group, relevant to your population, that includes patients, family members, and professionals; establish a PSP steering group; develop a protocol; gather uncertainties using national surveys; search existing literature to verify uncertainties; hold a final consensus workshop; produce a final report; disseminate and publish the priorities; and taking these to research funders. Naively, on completion of our first JLA PSP,2 we thought "job done" at this point. Our dissemination was exemplary, with a roadshow of conference presentations, we published,3,4 we evaluated parts of the process,5 and offered up, what we thought, was an excellent resource to our cancer community. We really believed that following this well-recognized consensus-building activity, we would enable research communities and funders of research to have greater confidence that they can address frontline priorities for future research. We were so confident in this process that we embarked on a second JLA (https://www.cclg.org.uk/news/top-10-priorities-for-childhood-cancer-research-revealed); this we completed in November 2022. What we know now that we did not know when we embarked on this road is that having confidence in a process, and making available to researchers and funders priorities for future research that have been identified by patients, carers, and professionals who care for them, is not enough. Readers might recall an editorial in Cancer Nursing,6 where there was a call for research to answer questions and uncertainties that are relevant to our patient populations. Two questions were highlighted in that editorial: (1) How far are the priorities of PSPs reflected in the research portfolio of the relevant funding bodies? (2) How far are researchers influenced by the priorities from PSPs? In this editorial, we reflect further, by asking an additional question: (3) What are the consequences of not responding to the results of PSPs, in this case, children with cancer? As identified already, the relationship between generating research priorities and people acting upon them is complex.7 We recognize that and recognize also our role in championing the Top 10, working with the Children's Cancer and Leukaemia Group, who funded our PSP, as well as other funders/stakeholders, to achieve greater success and impact. Therefore, what are our concerns, what continues to bother us? That is what we want to share with you here; these are our top 2 concerns. First, our primary concern surrounds the involvement of families in the process. In our recently completed PSP, we placed huge efforts on the inclusion of children. We know that the involvement of children and young people has not been easy to achieve. Certainly, Postma8 describes pediatric priority setting studies including a range of only 1-108 children. Seventy-one children (aged 3-21) from families impacted by childhood cancer, sent in 252 questions to our initial survey, and we held a workshop with 8 children (aged 8-16) to prioritize these questions. That is 79 children and young people who bothered to tell us what matters to them, thinking that we might seek to do something about it. Are we in danger of raising their expectations? Important here is that we ask children, and we listen, but action must also follow. The consequence of no-action is that trust might be lost in the process, and those in our care feel that what matters to them is not important, or maybe not as important, or valued by those who might choose to fund research. Second, there are the questions themselves. The Top 10 are disseminated widely, and no questions are ever lost. We had 1299 questions in our initial survey. Irrespective of the evidence checking, there is a lot of uncertainty in children's cancer care, shared by families and professionals. Let us just look at the Top 5: Can we find effective and kinder (less burdensome, more tolerable, with fewer short- and long-term effects) treatments for children with cancer, including relapsed cancer? Why do children develop cancer (including the role that genetics plays) and could it be prevented? Are the psychological, practical, and financial support needs of children with cancer, survivors, and their families being met during treatment and beyond? How can access to this support be improved and what further support would they like? How can we speed up the process of getting diagnosed and starting treatment in the right place? Why do children relapse, how can it be prevented, and what are the best ways to identify relapse earlier? Who would not want to fund these? Of course, they need refining, and yes, some might benefit from being framed as PICO (population-intervention-comparison-outcome),7 but these questions have survived a rigorous and transparent process and voted to reach the top. Would researchers in children's cancer care not want to study what was important to the end users of health research? The consequence of not seeing merit in these questions might be to endorse the continuation of research mismatch between patients and clinicians and researchers. Mirroring our first concern, there are consequences of no-action, contributing to an increase in research waste.9 Researchers, clinicians, patients, carers, and funding bodies all have a significant role to play in setting a research agenda, and then ensuring that research priority exercises do indeed shape that agenda from priority to funding. Contributing to building the evidence base to resolve uncertainties and make a meaningful difference in the lives of children and young people with cancer is our shared endeavor. But there are, we believe, consequences for not embracing the opportunities presented through these research priority exercises; implementing the research priorities, no matter how challenging it might first seem, must be the principal we all uphold. Acknowledgment Thank you to the Children's Cancer and Leukaemia Group and The Little Princess Trust for funding the children's cancer PSP.