Establishing the values for patient engagement (PE) in health-related quality of life (HRQoL) research: an international, multiple-stakeholder perspective

Patient engagement (PE), or patient and public involvement (PPI), is increasingly viewed as a cornerstone of health-related research activities and policy making [1]. Effective patient engagement (PE) can profoundly change how patient-centred research is conceptualised and conducted, resulting in better patient-centred care, management and measurement [2‐4]. However, these partnerships require new skills and sustained efforts for all stakeholders: understanding the values that different stakeholders aspire to provide an essential foundation for effective PE.

The values associated with good PE in health and social care research have recently been defined as ‘the established collective moral principles and accepted standards of a person or a social group; principles, standards or qualities considered worthwhile or desirable’ [5]. In supporting patients and health professionals to participate in effective PE, an agreed statement of values endeavours to support everyone in understanding their role—why we do it, what is important, and to whom. Experience has shown that different stakeholders often hold different values associated with the practise of PE; such discrepancies in values can result in conflict and a failure in the conduct of effective PE and its likely impact [5, 6]. In developing our understanding of the diversity in values, we can acknowledge and understand them and work within a framework that considers different perspectives, different motivations, and recognises the potential for conflict to emerge when such diversity exists. Developing strategies for managing such potential conflict are essential, highlighting the importance of understanding values at the outset of a programme of research and identifying common values that everyone recognises as well as respecting the diversity of values that may be present in a research team. A consensus process involving members of the Health Technology Assessment International (HTAi) Patient and Citizen Involvement Group (PCIG) recently defined five core values and standards to capture common understanding with which to underpin effective PE in HTAi processes: (1) relevance, (2) fairness, (3) equity, (4) legitimacy and (5) capacity building (http://​www.​htai.​org/​interest-groups/​patient-and-citizen-involvement/​pcig-home/​values-and-standards.​html) [7]. However, such explicit value statements for PE are rarely stated [5] and have not been explored for health-related quality of life (HRQoL) research.

The active engagement of patients as research partners is increasingly viewed as essential to ensuring that the patient perspective is considered throughout the research and healthcare process that research focuses on issues of importance to patients and that research waste is avoided [2, 3, 8, 9]. However, guidance for active PE in HRQoL research does not exist, and the evidence-base is limited, primarily because of poor reporting [10]. Moreover, the underpinning philosophical values held by different stakeholders may affect the PE approaches adopted and its likely impact. The results of the first International Society of Quality of Life (ISOQOL) research PE World Café [4] highlighted the need to agree and promote best practice for PE in patient-reported outcomes (PRO) and HRQoL research that is suitable in different healthcare and political systems. This can be achieved through the creation of a set of values and high level principles to support best practice, developed with wide stakeholder engagement and in a scientifically robust manner to ensure use in practice [5, 10, 11].

As a continuation of ISOQOL’s commitment to embracing PE in measurement science and HRQoL research, in October 2014 it hosted a second PE World Café with the intent to develop clarity and understanding of PE/PPI with an international audience of patients, healthcare professionals and researchers. It was envisaged that this would lead to an international consensus on a set of values and quality standard statements for PE/PPI in the development and use of HRQoL measurement.

This study provides the first international exploration of values that should underpin PE in measurement science, HRQoL and PRO-related research, reflecting the perspective of patients, healthcare professionals, researchers and academics from healthcare, policy and industry. The result is an explicit statement of three core values that seek to align all stakeholders on the purpose, practice and credibility of PE activities. Participants valued the importance of building genuine, collaborative and deliberative relationships between all stakeholders, underpinned by honesty, respect, co-learning and equity. Also valued was the impact of effective PE on the quality, relevance of research, and the implementation of research findings. An innovative, flexible and transparent research environment was also valued as essential to developing a trustworthy evidence-base with which to underpin future guidance for good PE practice.

In seeking to co-produce a value statement for PE, reference was made to the Public Involvement Impact Assessment Framework (PiiAF) [6]. This framework was informed by an extensive programme of research that sought to define the values associated with best practice PE/PPI in health and social care. The framework was provided as an aid to study participants to guide their deliberations. However, the framework received a mixed response. Whilst some participants found it useful, others found it challenging, and choosing not to refer to it. Nevertheless, the PiiAF provided a useful basis to inform the data analysis. Indeed, the three proposed values clearly mapped to two of the PiiAF value systems and most of the associated cluster values: specifically, values associated with the process and consequence of PE/PPI.

Participation from North America and Europe at the ISOQOL 2014 conference was well balanced, and this mix was similarly represented in the PE café event. Although, as expected, the number of health professionals outnumbered those of patient partners, almost 20% of the group were patient partners. This mix enabled between one and two patients per table discussion, facilitating a real conversation between patients and the ISOQOL community and thus enhancing confidence in the external validity of the results. Patient research partners contribute approximately 10% of OMERACT conference participants—a group that, over the last decade, has championed the active engagement of patients as research partners in PRO-related research [2, 13]. Moreover, both the European League Against Rheumatism (EULAR) (http://​www.​eular.​org/​pare_​patient_​research_​partners.​cfm) [18, 19] and OMERACT [20] have recommended the inclusion of at least two patient partners per research project; this is a recommendation that should be discussed further within the ISOQOL community.

The patient partners represented a diversity of health conditions and experience of engagement as research partners. Although all patients were sponsored to attend the event and hence may have provided a more positive experience of PE, the task of exploring values associated with PE was novel to all participants. Specific data on health professional participants were not captured, with participants self-selecting to participate in the event and thus were likely to represent a mix of ideas and experiences (both positive and negative). In keeping with the ethos behind good patient engagement [18‐20], the ‘PE theme team’ sought to facilitate a collaborative approach to the group discussions and grounded in mutual respect for the perspective of others. Hence, an important learning point was the change from having patients participate as a single table to integration across all tables. The original decision was driven by a desire to understand whether patients and health professionals valued PE differently. However, it was clear that the opportunity for discussion amongst all participants, leading to a transparent, co-production of values was more highly valued by participants.

This is the first study to describe the use of a ‘World Café’ type format to explore the values associated with patient engagement in HRQoL research. The format was well received by all participants—particularly once groups were ‘mixed’—who welcomed the opportunity to openly engage with colleagues within a mutually supportive environment. However, participants were set a significant task that, on reflection, would have benefitted from longer periods for discussion and reflection—as reflected in more traditional World Café approaches [12]—than possible within the available 90 min.

In addition to the recent HTAi PCIG values statement, there are helpful similarities between the values proposed by this study and those of several other groups. OMERACT recognises that effective PE strengthens research; it therefore actively promotes the equitable participation and integration of patient research partners with professional researchers throughout all OMERACT projects [13] (http://​www.​omeract.​org/​pdf/​OMERACT_​Handbook.​pdf). The European Patients Forum have defined five values to underpin meaningful patient involvement: (1) appropriate patient representation; (2) building on diversity and pooling knowledge; (3) equality, providing an empowering environment; (4) commitment to patient involvement as a positive, value-adding experience; and (5) respect for patients as equal partners [21] (European Patients Forum: The Value+ Toolkit: http://​www.​eu-patient.​eu/​globalassets/​projects/​valueplus/​value-toolkit.​pdf). These values are also embraced by the Patient Centred Outcomes Research Initiative (PCORI), where the principles of trust, honesty, co-learning, transparency, reciprocal relationships, partnership and respect underpin their stated approach towards patient-centred outcomes research [8] [http://​www.​pcori.​org/​about-us]. The importance of building strong relationships between all stakeholders which facilitates the active involvement and contribution of all members is central to most of these shared values and is mirrored in the recommendations from the ISOQOL PE café participants. Moreover, the importance of establishing and maintaining good relationships between researchers and lay representatives has recently emerged as a key aspect of collective action—the operational work required to enact PPI practices [22]. This was done through regular communication, managing meetings to address power imbalances and providing opportunities for informal engagement. However, there was some evidence that developing good relationships was difficult when PPI was conducted purely through virtual media [22, 23].

Growing evidence suggests that meaningful PE requires an engaged research team with a shared understanding of the collective values and an awareness of the sustained, long-term effort that should underpin PE. However, additional elements that may facilitate more effective PE moving forward are as follows:

Methodological guidance—developing evidence-based guidance for how to do it and what works PE is far more than supporting patient participation in interviews or focus groups. Rather, PE/PPI is a ‘collaborative partnership between patients and researchers, which is often underpinned by the intent to co-produce knowledge’ [10]. In HRQoL research, such active engagement can, for example, support the production of more patient-relevant research questions, enhance study design so that it is more acceptable and appropriate for patients and inform outcome selection so that the outcomes that really matter to patients are the focus of the research and hence avoid the potential for research waste [9]. Moreover, such engagement can support the selection of measures that are both robust and relevant.

As with many new areas of research, the reporting of PE activities is poor. In order to develop a strong evidence-base that benefits future PE research, the transparency and quality of reporting must be improved. Moreover, the involvement of patient partners as co-authors in published research, and the inclusion of a section which communicates the conduct and value of research to a lay, non-academic audience are increasingly recommended (http://​www.​bmj.​com/​campaign/​patient-partnership). The GRIPP 2 (Good Reporting of Involving Patients and the Public in research) [10] initiative has provided the first international evidence-based, consensus informed checklist for PE (PPI) reporting. The application of such a framework in HRQoL and PRO-related research has yet to be explored: extending GRIPP 2 for HRQoL could support a developing evidence-base through better reporting, facilitating a clearer evaluation of PE. Once methodological guidance is established, a logical and essential next step is to define the measurable standards—the elements of best practice—against which good practice will be assessed.

Tailored education and long-term support for both patient partners and other members of the research team to enable full participation, communication and engagement throughout all stages of the research process and beyond. Access to a developing evidence-base of good practice, or a registry of PE activities and initiatives, could support all researchers, including patient research partners, in developing their practice, skills and aptitudes to become effective PE practitioners. For example, experience from the OMERACT group highlighted the importance of developing a network of patient partners with a range of experiences who can offer ‘buddy’ support for newer and less experienced patient partners in the process [13, 19, 20]. However, as evidenced by groups such as OMERACT [13], EULAR [20] and PCORI [8], whilst such activities are essential to establish and maintain effective research partnerships, they are resource intensive—in terms of time and cost.

Supportive institutions that value the contribution of PE whilst the financial requirements of effective PE are increasingly recognised by grant awarding bodies, the lack of institutional support has been reported as a barrier to PE [4]. The additional, and often upfront, costs of PE throughout the research process—including the appropriate training of patient partners and researchers—must be recognised and appropriately budgeted for (http://​www.​pcori.​org/​sites/​default/​files/​PCORI-Budgeting-for-Engagement-Activities.​pdf). Supportive institutional policies, underpinned by reference to core PE values such as those established in this study, are essential when seeking to develop a strong and sustainable foundation for PE activities.

In providing the first, international and multi-perspective statement of values for PE in HRQoL, measurement science and PRO-related research, this study provides a strong foundation for the alignment of future PE activities and good patient engagement practice. The wider adoption of these values will facilitate a more transparent understanding of the importance of PE in this field.