Patient and researcher perspectives on facilitating patient and public involvement in rheumatology research (2016)

The Arthritis Research UK Rheumatoid Arthritis Pathogenesis Centre of Excellence (RACE) is a partnership between researchers at Newcastle University, the University of Birmingham and the University of Glasgow. Established in 2013, it is funded for 5 years, with a grant of £2.5 million from Arthritis Research UK and a further £4 million pledged by the three universities. Rheumatoid arthritis (RA) is a chronic inflammatory autoimmune condition that affects the joints and internal organs. Synovial inflammation can cause cartilage and bone damage with resultant joint destruction and associated disability. RACE aims to identify biological mechanisms involved in the initiation of RA and its progression to a chronic disease, and to identify novel therapeutic targets for control and cure. PPI in all stages of the research process is advocated by funding bodies and policy makers as a way of enhancing the relevance, quality and efficiency of research (INVOLVE, 2012; National Institute for Health Research, 2015). The input of PPI partners has been shown to enhance the design of clinical trials (Brett et al., 2014), development of patient-relevant research ideas (Bergsten et al., 2014), agenda setting (De Wit, Abma, Koelewijn-van Loon, Collins, & Kirwan, 2013) and developing appropriate patient-reported outcomes for clinical trials (de Wit, Kvien, & Gossec, 2015). Evidence suggests that patients identify different treatment priorities to clinicians (da Silva et al., 2010; Kwoh & Ibrahim, 2001). This reinforces the case for involving patients in the design of research to ensure that their priorities are addressed, and to improve the quality and impact of that research. Patients have been involved in rheumatology research at each of the three hubs prior to the inception of RACE; therefore, the project offers a unique opportunity to bring together patient partners from each of the hubs, to share experiences and best practice. Details of these groups can be found in Table 1. Members of R2P2 are actively involved in all aspects of the research process, including the development of grant applications; the design of study procedures and of participant-facing research materials; the development of informational resources; and the dissemination of research findings via patient networks and support groups. Patient partners are co-authors on papers, including those related to the development of international research recommendations (Gerlag et al., 2012); the development of patient questionnaires (Stack et al., 2015) and patient-initiated research to determine why people with symptoms of rheumatoid arthritis delay seeking help (Tiwana, Rowland, Fincher, Raza, & Stack, 2015). The conference was attended by two patient research partners from Birmingham, five from Glasgow and six from Newcastle, 11 of whom completed a questionnaire prior to the conference, to describe their existing experience of research involvement. The participants' previous involvement in research activities is summarized in Tables 2 and 3. Patient partners who attended the conference had limited previous research involvement but were enthusiastic to become involved, as evidenced by their attendance and comments at the conference. Respondents were also asked to describe any difficulties they had experienced or anticipated from PPI in research. Many had encountered or anticipated no difficulties, although some raised concerns about the time commitment required, the use of technical language or the possibility that deterioration in their health might result in them withdrawing or reducing their involvement. Clinicians and researchers from each hub were also present, along with the RACE project manager and a representative from Arthritis Research UK. Following an introductory presentation on PPI and the existing contribution of patient partners to rheumatology research at each hub, delegates split into groups to facilitate discussion and debate of the main topic areas. A third group of scientific researchers and clinicians affiliated with the University of Birmingham met separately to discuss the same questions. The three groups were then brought together to share key discussion points, which are summarized below, under each topic. There were some key differences between the views expressed by patient representatives and the expectations of researchers and clinicians. The latter group felt that patients would wish resources to be directed towards development of new treatments, particularly for those with longstanding, difficult-to-treat RA, who are increasingly described as becoming a neglected group, in terms of research priorities. The researchers/clinicians anticipated that patients' priorities would feature research into the effectiveness of non-pharmacological approaches for the management of RA. The patient group agreed that there was a need for evidence of the effectiveness of such approaches. Patient representatives also reported that a focus on the therapeutic effectiveness of treatments was more desirable than a focus on their cost-effectiveness. The importance of PPI in research, and research agenda setting, was agreed by all three discussion groups. The challenges of involving patients in basic laboratory-based research were also discussed, with particular reference to the language gap between patients and researchers. Examples of successful approaches to this problem in previous projects were shared, such as the development of a glossary for PPI partners taking part in the European Union's FP7 project “EuroTEAM” (Towards Early diagnosis and biomarker validation in Arthritis Management: www.team-arthritis.eu). Many patient partners perceived opportunities to become involved in research to be limited, and reported a lack of public awareness of such opportunities, and of the benefits of becoming involved. Benefits that were suggested included the ability to access the latest research findings and to engage with researchers, as well as the opportunity to “give something back” for care received by contributing to the research process. It was suggested that access to such benefits should be publicized and routinely available to all patients. A concern was raised by patients that those who become involved with research may not be representative of the wider patient group, and often develop extensive research expertise, leading to a possible loss of perspective. It was agreed that it would be important to identify ways to widen access to research involvement in order to mitigate this. Patient representatives commented that it is not always straightforward for patients to attend research meetings in person, and opportunities to learn about involvement and contribute to research in other ways (e.g. via email or Skype) were welcomed. The clinicians and researchers acknowledged the variety of opportunities for involving people throughout the research cycle (Figure 1), and highlighted the responsibility of researchers to be aware of the benefits of PPI, and to provide patients with comprehensive information about current research and future possibilities in order for them to be able to make fully informed suggestions and choices about research priorities. It was suggested that, in this way, the research cycle (Figure 1) should be seen as a virtuous cycle, where effective dissemination of research findings to patients facilitates the input of patients to the research agenda, and their ongoing engagement with the research process. This was echoed by patient representatives, who suggested that patient involvement should be an important aspect of training of researchers, including those involved in basic, laboratory-based science. All groups agreed that patients could usefully be involved in the development and distribution of resources to communicate research findings. Furthermore, it was felt that groups such the Birmingham Rheumatology Research Patient Partnership (R2P2), the Glasgow Patient Involvement in Rheumatology Research (PIRR) group and the Newcastle Public Involvement in Musculoskeletal Services (PIMS) group, or other patient-organized groups were an important mechanism for the dissemination of research findings. It was suggested by patient representatives that it might be helpful to conceptualize three tiers of target audience: patients already actively engaged with research; patients who are not engaged; and the wider public. Thus, dissemination to local groups who can then cascade information via existing networks was perceived to be a useful approach. There was a strong consensus that this type of dissemination activity needed to occur at regular intervals, with a well-publicized timetable. Patient representatives involved in local groups suggested that they would value a database of local researchers who were willing to present their work to patients, along with opportunities to link with websites or mail-outs associated with other local and national patient groups. While the importance of increased opportunities for social networking in this context was discussed, patient representatives felt that traditional media should not be neglected, as many patients do not use the internet or social media. Several patients suggested that patient-friendly posters displayed in outpatient settings could be an effective route for local dissemination. In the researchers' group, there was discussion about whether clinicians or scientists were best placed to disseminate research findings to patients. Some concern was expressed by non-clinical researchers that there would be a perception on the part of patients that all researchers would have clinical knowledge. However, several non-clinical researchers described very positive experiences of face-to-face dissemination to patient groups, although it was acknowledged that preparatory work was necessary to ensure appropriate use of non-technical language and management of patients' expectations in relation to the researcher's ability to answer clinical questions. Patient representatives stressed the importance of communicating with patient partners throughout the research cycle, as well as on completion of specific research projects, and highlighted the need for training for researchers from an early stage of their career, to provide them with skills in the communication of research findings to patients and the wider public. It was suggested that university press offices could be better engaged with scientists, and that there was a potentially useful role for patients to be involved in the identification of tractable items arising from research findings that might be seen as newsworthy. In summary, there was wide agreement about the benefits of PPI in rheumatology research for all stakeholders. It was also generally agreed that research into the basic disease mechanisms of RA and prediction of disease outcome should be important priorities, although scientists/clinicians had expected patients to place greater emphasis on the development of new treatments and non-pharmacological approaches. Participants shared their experiences of dissemination activities and identified effective strategies and opportunities for patient involvement in this context. An important role for patient groups and networks was identified in this context. We acknowledge that participating patients had previously expressed an interest in involvement with the RACE project, and their views may not represent those of all patients with RA. Further research is necessary to ascertain variations in patient perspectives in this context. The authors wish to thank the patient representatives who attended the RACE patient conference and provided the basis of this work. This work was funded by grant number 20298 from Arthritis Research UK.