What do young people with rheumatic conditions in the UK think about research involvement? A qualitative study

Involving people at all stages of health research (including young people) is now widely recommended [1]. In the UK, many grant and all ethics application processes now ensure that patient and public involvement (PPI) in research is embedded within them [2]. PPI is also advocated by professional bodies in general and specifically for young people. [3, 4].

Within this study we defined involvement as “where members of the public are actively involved in research projects and in research organisations” [5] and participation as, “where patients and the public act as research participants”.

Involving patients and the public in research helps ensure that research is designed around their needs and what is important to them [6]. However, it can be challenging to involve a diverse range of people [7]. This is particularly true with young people [8]. In view of the major sociocultural influences on adolescent health [9], representative sampling (in terms of socio-demographics- gender, ethnicity, culture and urban vs rural regional variations) is vital in studies of adolescents in general and when not feasible, the limitations of non-representative samples should be stated. In view of the wide variation of normal puberty, chronological age is a poor indicator of developmental status. Therefore, attention to the stages of adolescent development is required in any research involving young people. Ensuring such diversity may also result in part from a lack of confidence and/or inexperience amongst researchers regarding PPI [10], particularly with respect to young people in these particular developmental stages as although clinical research focusing on young people is rapidly evolving, it is still developing compared to research with adult populations [11].

The Barbara Ansell National Network for Adolescent Rheumatology (BANNAR) is a network of rheumatology professionals aiming to ensure that young people in the UK have the best chance to benefit from developments in the field of adolescent and young adult rheumatology [12]. A key priority for BANNAR is to involve young people in developing the network’s research priorities.

This study aimed to explore (i) experiences of research participation and involvement (ii) beliefs about research involvement amongst young people with rheumatic conditions and (iii) beliefs about how young people’s involvement should be organized in the future.

Data regarding their research priorities has been published elsewhere [13].

This was a qualitative study of young people with rheumatic conditions. Sixteen focus groups were planned (8 with 11–15 year olds and 8 with 16–24 year olds) in all four nations of the UK to capture the potential impact of differences in health service organisation on young people’s experiences. The age ranges were chosen to reflect adolescent developmental stages i.e. early and mid-adolescence (11–15 years) and late adolescence and young adulthood (16–24 years).. Sixteen focus groups were conducted in all four nations of the UK to capture the potential impact of differences in health service organisation on young people’s experiences. The methods for this project have already been reported in detail elsewhere [13, 14].

Thirteen focus groups were held across the UK (England 8; Scotland 2; Northern Ireland 2; Wales 1). The original aim had been to conduct 16 focus groups but data saturation was achieved after 11. We determined that no new ideas were generated via reviewing recordings and transcripts and early analyses of the data. We however conducted a further 2 focus groups to ensure that young people from all four nations of the UK had the opportunity to participate although no new ideas were identified in this groups.

Six groups were held with 11–15 year olds (n = 30) and seven with 16–24 year olds (n = 33). Participants’ ages ranged from 11 to 24 years (mean = 16), 20 of whom were male and 43 female. Characteristics of participants are detailed in Table 1.

The themes which were identified are detailed in Table 2.

To the best of our knowledge, this is the first study, to explore the experiences and beliefs of young people with rheumatic conditions about research involvement. Although young people felt that they had a right to be involved in research, experience of research involvement varied. This may reflect geographical variation in the research culture, i.e. the extent to which research participation and involvement of people in research were viewed as priorities and key elements of usual care by clinicians, researchers and their host institutions.

Young people also expressed a wish to increase their research awareness. This may have arisen in part due to difficulties in remembering whether they had taken part in research especially if what was involved was similar to usual care e.g. completing the Childhood Assessment Questionnaire [17].

If young people believe that they have a low awareness of research in general, then it is also likely to be difficult to increase their involvement as a public contributor. In this study some young people reported never being approached about research participation nor research involvement. This concurs with the findings of a previous UK audit of paediatric and adolescent rheumatology services which reported that at least 60% of centres did not involve young people in research other than as research participants [14].

An interesting finding was that some young people who had actually been involved in shaping research did not always perceive this as involvement. One explanation for this may be the lack of feedback they received following their involvement, or it may reflect lack of clarity of their role during the participation and involvement processes.

A key strength of this study was the inclusion of 63 young people from a broad range of ages and from all four UK nations, thereby reflecting a range of service provision and research opportunities. The variation in the experiences of research involvement and participation was an important finding, validating our approach and highlighting the need to conduct a national study. However, recruitment for this study was poor in some areas particularly if there was not a PPI or transition coordinator who could help with recruitment. In the development phase of this research, a survey of the 25 main paediatric rheumatology units identified that just five units had a team member with PPI included in their job description [14]. Furthermore, in order to limit recruitment bias, we adopted a maximum variety purposive sampling approach to sampling for this study. This aimed to encompass the range of AYA development by having focus groups for the first 2 stages (early and mid) and the latter 2 stages (late adolescence and young adulthood). We also involved centres in all 4 devolved nations of the UK in order to capture the range of services for this age group, in both urban and rural settings. There has been a significant growth in specialist services for this age group in recent years in the UK although significant delays in referral persist for certain conditions eg juvenile idiopathic arthritis [18]. To further maximise the diversity within our recruitment we recruited via charities in an attempt to recruit young people not being treated at centres with specific paediatric, adolescent and/or young adult rheumatology services. These young people potentially had less access to research opportunities.

However despite efforts to recruit a maximum variety sample, due to the relative small sample sizes required within qualitative research there will still be some limitations in terms of the diversity of the sample. For example, research-naïve young people or those with very mild disease and/or who are happy with their current care may not have perceived a study focused on beliefs about research involvement as being relevant to them. However we did successfully recruit such young people in this study. Another group less likely to engage in research are those young people who have become disengaged from their rheumatology care. Understanding their perspectives would be particularly valuable. Reasons for non-response are important in any research but particularly pertinent to adolescent health as some studies have shown that those who fail to respond have poorer outcomes compared to adult non-responders [19].

An important impact of this study is that, incorporating the clear advice regarding involvement in the current study, we have since established a national young person’s advisory group to inform BANNAR research.

The group is called Your Rheum (https://​yourrheum.​org/​) [20] and is currently holding face to face meetings several times a year and involving young people online in research.

Finally, whilst acknowledging the exploratory nature of the current study, one could argue that many of these findings are also true for adult populations. However, implementing change in the adolescent and young adult group in relation to involvement, just as in clinical care, requires developmentally appropriate approaches which change over time as young people grow and develop [11].

Young people identified a lack of opportunity or a perception of poor access to research and research involvement as a primary barrier to research involvement. This suggests that efforts are needed to increase researchers’ awareness and understanding of PPI and its’ likely impact. Increased awareness may increase researchers’ confidence in involving young people in their work and lead to a wider range of research involvement opportunities becoming available. As part of this current project, models of good research involvement practice in this area beyond rheumatology were collated to serve as a future resource for researchers [21]. Since the Clinical Studies Group in Paediatric Rheumatology was established [22], research involvement outside of large teaching hospitals has significantly improved but further work is still required, if we consider the views of participants in the current study of their poor access to involvement opportunities. Investment into appropriate staffing for such initiatives is supported by the finding that recruitment to this particular study was better in those centres with a team member with PPI as part of their job description.

The findings from this exploratory study also suggest that further work is needed to increase young people’s awareness of rheumatology research within the UK. The national paediatric rheumatology clinical studies group, which supports a portfolio of clinical studies across the UK states:

“All children and young people in the UK with a rheumatological condition may be given the opportunity to be enrolled in a clinical trial or well conducted clinical study from point of diagnosis onwards” [22].

The current study has revealed that it will be important to ensure both that the aims and purposes of young people’s involvement in research are made clear to them as well as receiving feedback on both their involvement as well as their research participation. It will also be important to explore the language used to explain research participation and involvement to young people to gain some insights into why the nature of involvement is sometimes misconstrued.

In this current study, few young people had experience of being involved in influencing research, with those treated in large teaching hospitals being more likely to report having experience. Despite this finding, all young people strongly believed that they should be involved in research, particularly as they had lived experience of their condition and could provide a perspective which would otherwise not be available. Acknowledgement of the lived experience of young people with rheumatic disease is therefore as imperative for researchers as has also been reported for clinicians [23].

Understanding and evaluating the impact of patient and public involvement in general, and for young people is becoming an increasingly important issue. Evaluation frameworks for involvement have been developed for adults but it is still unclear whether they can be used effectively with young people [24]. The involvement of young people in research has been reported to have a positive impact on recruitment and retention [25]. However, Holland et al. cautions practitioners “against assuming that participatory research per se necessarily produces “better” research data, equalises power relations or enhances ethical integrity” [26]. Therefore, research is needed to explore how young people perceive their roles as active research partners in the context of chronic health conditions when involvement could potentially be an additional burden [27].

This exploratory study highlights the importance of further enhancing the culture of research in the adolescent and young adult age group, to increase young people’s awareness of opportunities for both research participation and research involvement. In the UK, BANNAR and the YOURR project have made initial steps in doing so in rheumatology.