Preparing for patient partnership: A scoping review of patient partner engagement and evaluation in research

1Including patient partners in research holds promise for targeting patient‐important research questions, creating meaningful change in patient outcomes and health systems, and realigning both research processes and outcomes to be patient‐centred.1 Internationally, efforts to grow inclusion of patient representatives on research teams have been driven by funding bodies, government and calls to action by patient communities.2, 3, 4 Patient partnership represents a growing niche in the broader field of patient engagement, whereby patients take on a more collaborative than contributory role in the research process. Consistent with definitions from the Patient‐Centered Outcomes Research Institute (PCORI)5 and the Strategy for Patient Oriented Research Patient Engagement Framework (SPOR),6 the term ‘patient partners’ within this manuscript is intended to include patients and their proxies (ie family members or caregivers) who are directly involved as members of the research team, as opposed to being consenting research participants. In this study, this definition was operationalized by only including patient partners who were not consented as study participants, but members of the research team. On the continuum of patient engagement practices described by Forsythe and colleagues,4 as well as the Levels of Patient and Researcher Engagement in Health Research proposed by Manafo et al7 patient partnerships are typically those that involve collaboration, shared leadership practices and patient partner embeddedness in research teams as co‐investigators. In contrast to broad engagement practices which commonly involve unidirectional input from patients to researchers via solicitation of viewpoints or experiences to inform the research agenda, patient partner relationships within research teams are characterized by bidirectional information flow and active decision making and collaboration.4

2Historical efforts aimed at involving patients in research began with broad engagement of various stakeholder groups. For example, within the Canadian health‐care system, the Canadian Institutes of Health Research (CIHR) 2014/15‐2018/19 Strategic Plan, Health Research Roadmap II (HRR‐II), focuses on mobilizing health research for transformation and optimal impact on the health of Canadians.8 To meet this challenge, the HRR‐II calls for scientific leaders to employ a highly networked approach, inclusive of multiple stakeholder groups, in order to transcend traditional strategic alliances for health research. A key priority for such collective action is partnership with citizens, patients and caregivers with a view to effectively accelerate innovation, and ultimately create better health and health care. Similar initiatives have been fully launched outside of Canada including INVOLVE, funded by the National Health Service in the United Kingdom, and the Patient‐Centered Outcomes Research Institute (PCORI) in the United States.

3A primary driver in the field of patient engagement is active collaboration with patients on research teams – including family, caregivers and friends – in governance, priority setting, conduct of research, and knowledge translation.9 However, many researchers continue to struggle with how to operationalize research partnerships with patients, both realistically and effectively. We propose that partnerships require a shift in focus from engaging research participants in ancillary roles to more active roles, wherein patients engage as collaborative team members throughout the research process. While patient partnerships are often discussed, their impacts are rarely measured.10 Previous systematic reviews2, 3, 11, 12 on the involvement and engagement of patients in research have not differentiated patient partners from consenting research participants who are engaged in research. A knowledge gap exists related to whether patient partnerships – as a subset of patient engagement – have an impact on research outcomes.

4The primary aims of this work were as follows: (a) to identify the body of research where patients were included as patient partners in the research process; and (b) to determine whether the impact of patient partnership was examined and, if so, what impact the partnership had on research outcomes. The secondary aim was to describe specific partnership methods, barriers and facilitators across included studies in order to make practical recommendations about operationalizing patient partnerships.

5The scoping review was guided by the Arksey and O'Malley Scoping Review Framework13 which lends structure for identifying the research question and relevant studies, selecting studies, charting the data, and collating, summarizing and reporting results.

6Within the existing body of health research literature, how is the impact of patient partnership being examined and, when examined, what impact does partnership have on research outcomes?

7The search strategy was developed with an experienced information scientist in consultation with the research team. The search was undertaken in Ebsco CINAHL, and EMBASE, MEDLINE and PsycINFO via Ovid. No limits for language, date or publication type were applied. Using a combination of keywords and database‐specific subject headings, the concept of community‐engaged/patient partnership research (encompassing community‐institution relationships and patient or consumer participation) was constructed as a concept for this search. This concept was then combined with language describing health services research or research design to form the basis of the search strategy. All databases were searched from database inception to October 2019. For a sample MEDLINE search, see Appendix 1 (other search strategies are available on request). Once duplicates were removed, citations were uploaded to a web‐based program (Distiller SR)14 and the title and abstract of each citation were screened by one reviewer. In addition, reference lists of included articles were hand‐searched for possible additional articles.

8The population of interest was defined as patients or their proxies including informal caregivers, family and/or friends who were considered patient partners during any stage of health research. Only studies where patient partners had not signed consent were included in this review as we interpreted the practice of consent to imply a research participant role versus a partner in the research process. All quantitative, qualitative or mixed methods peer‐reviewed research articles from any health‐care setting were included. Included articles described an evaluation of the impact of patient partnerships on research outcomes using validated tools for quantitative studies, or patient partner interviews or focus groups for qualitative studies. This applied examination of the impact of patient partnerships necessarily excluded studies where the objective was to develop a measure or tool to evaluate patient partnership, for example (see Appendix 2 for Screening Criteria).

9At the initial screening level, both titles and abstracts of citations were screened by a single reviewer. Full‐text screening was conducted independently by two reviewers, with agreement necessary for inclusion. Disagreements were reconciled through discussion prior to exclusion of studies.

10Data extraction from included studies was independently completed by two reviewers. All disagreements were resolved through consensus between reviewers. Study characteristics were extracted, as well as process for patient partner involvement, patient partner role/contribution, barriers and facilitators to partnership, approach to evaluation, and ‘stage’ of patient engagement (Manafo et al7). Data were extracted using a standardized data extraction form. See Figure 1 for flow of studies through the phases of selection, data extraction and synthesis, according to Preferred Reporting Items for Systematic Reviews and Meta‐Analyses (PRISMA) guidelines.15

11Results were summarized using a narrative synthesis process consistent with Arksey and O’Malley's Framework.13 Our approach to narrative synthesis followed guidance provided by Popay and colleagues16 and included a descriptive summary of study characteristics (including country of origin, research method, diagnostic focus and patient partnership method), and an exploration of relationships between studies' reported findings. Study findings were grouped into categories related to perceived patient partner contribution, as well as barriers/facilitators to partnership. Each included study was also assigned a level of patient engagement as proposed by Manafo et al7 in order to depict variations in the operationalization of patient partnerships across studies. Findings were described and synthesized across studies.

12The search yielded 31 184 unique citations which included 31 175 from the database review and nine studies from a previously published systematic review.12. After title and abstract screening, 29 897 citations were excluded, leaving 1287 for full‐text review. After full‐text screening, an additional 1273 articles were excluded, leaving a total of 14 included studies in this review.

13All 14 of the included studies utilized qualitative data collection and analysis techniques to evaluate the impact of patient partner engagement on research outcomes.17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30 Table 1 summarizes the key study characteristics. Eight studies originated from the United Kingdom, 18, 19, 20, 22, 23, 24, 25, 26 one from Canada,21 one from Sweden,27 and four from the United States of America.17, 28, 29, 30 The included studies used a combination of qualitative data collection methods, including interviews (n = 8),17, 18, 19, 20, 21, 24, 28, 30 focus groups (n = 8),17, 18, 20, 22, 23, 24, 25, 26 questionnaires (n = 5),22, 24, 27, 28, 29 reflections (n = 1)27 and observations (n = 7).18, 21, 25, 26, 27, 29, 30 The focus of five of the studies was on chronic conditions, including musculoskeletal conditions,26 diabetes,23 stroke,20 Parkinson's disease27 and asthma.30 Three of the studies examined populations with cancer,18, 19, 22 two were in varied populations,25, 28 one focused on the primary care setting,29 and another involve street‐involved youth.21 Additionally, one study focused on sexuality in older adults,24 and one study evaluated patient partnerships with adults with developmental disabilities.17

14In what follows, we describe data extracted from the included studies in two main areas: (a) contributions of patient partners and related examination of impact; and (b) practical considerations for creating partnerships, including barriers and facilitators influencing patient partnerships. As noted below, the depth of patient involvement varied across studies, with some studies engaging patient partners throughout the research process, and some engaging patient partners for specific tasks. The levels of patient engagement7 included in Table 1 highlight these variations. Most studies involved patients in more than one manner (ie Consult, Involve, Collaborate), and slightly more than half of studies demonstrated engagement with patient partners at the stage of ‘Collaboration’.7

15Across studies,17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30 patient partner contributions were described with respect to roles enacted and direct involvement in study‐related activities. The impact of partnership was largely reflected in terms of personal experience and gains.

16Patient partners in included studies enacted various roles within the research team. Table 2 provides an overview of these roles.

17Nine included studies described patient partners as members on governance structures within the research study. These governance structures included steering committees17, 19, 23, 25, 29, 30 and advisory boards.17, 18, 19, 20, 25, 28, 30 For example, one study17 included persons with developmental disabilities on the study steering committee as well as community advisory boards where partners provided input on the development of instruments and tools to assess accessibility for people with developmental disabilities. In two other studies, patient partners acted as advisors during the conduct of a systematic review,22 and as leaders in the planning of an annual conference,23 respectively. Patient partners in these studies contributed to study outcomes by advancing a research agenda that incorporated patient‐important outcomes.

18Eight studies described patient partners enacting consultant‐type roles in the research work.17, 18, 22, 24, 26, 28, 29, 30 This consulting role varied across studies and included sharing the lived experience of the partner, (eg sharing how the intervention would affect patients29) contributing to strategy related to the development of plain language and accessible study materials,17, 24 and consulting on patient‐centred knowledge translation strategies.30

19Further, four included studies involved partners as research co‐designers.18, 20, 27, 29 For example, in one study, four separate co‐design workshops were organized in order to solicit feedback from partners with Parkinson's disease to inform and explore care needs.27 In another study, twelve patient partners participated in a four‐day co‐design workshop with researchers to create a new health‐care service role.29 Co‐design activities with patient partners typically involved periods of intense meetings to share ideas and undertake creative activities with other research team members, sometimes followed by ‘check‐ins’ to ensure co‐designed products reflected the contributions of patient partners.27, 29

20In nine of the studies, patient partners were involved in knowledge translation and dissemination efforts in both formal and informal capacities.19, 20, 21, 22, 23, 24, 26, 28, 30 By virtue of being engaged, some patient partners became de facto resources for their communities regarding disease‐specific knowledge.22, 23 Additionally, and more formally, patient partners presented findings at project meetings22 and academic conferences,19, 21, 24 and were engaged in knowledge transfer activities such as networking with various health agencies and academic research partners,21 which lent credibility to study results in the eyes of stakeholders.

21Six studies discussed patient partners taking on specific research‐related processes and tasks,21, 22, 25, 26, 28, 30 which included the development of research questions,28 interview guides and informational materials,19 as well as identification of research priorities,21, 28 and contributions to data analysis28 and to regular study briefings.22 In one study using a participatory approach, youth co‐researchers interfaced directly with study participants by facilitating focus groups with street‐involved youth.21

22In addition to the contributions to research processes described above, personal benefits of patient partnership were also noted. For example, patient partners described acquiring practical skills19, 21, 25, 29 (eg learning to use a computer17) and gaining knowledge about research processes and various topics.19, 21, 22, 23, 29 Some patient partners reported that the process of collaborating with researchers helped them to gain confidence in identifying themselves as experts and advocates.17, 20, 23 Patient partners also shared that participation in research afforded them a social network of supportive peers; some noted that relationships forged – both personal and professional – lasted well beyond the study period22, 23 and that participation was a source of positivity (eg laughter)24 Finally, patient partners articulated that adding the ‘patient voice’ to research projects and advocating for change was an empowering experience.19, 22, 23 Because of these personal impacts, some studies reported that patient partners sought further opportunities to become involved in research as patient partners beyond study completion.20, 21

23Practical considerations were addressed largely in terms of barriers and facilitators to patient partnership. A summary of lessons learned, barriers and facilitators to patient partnership, as reported by authors of included studies, can be found in Table 3.

24Eleven studies cited specific barriers to partnership encountered during their work.17, 19, 21, 22, 23, 24, 25, 26, 27, 28, 29 These barriers were varied and encompassed study logistics, team characteristics and perceptions of patient partner roles by researchers, as well as partners themselves. One of the most common barriers identified was the use of jargon.19, 23, 24, 25, 29 In one study, both researchers and patient partners acknowledged that the nature of some discussion topics, such as prospective funding sources or ethics applications, made it difficult for patient partners to understand and follow what was being discussed.24 Researchers in this study reported feeling pressured to limit their conversations to ‘non‐academic’ topics, in order to limit the use of jargon.24 Other common barriers described included power imbalances between the researcher and the patient partners,17, 24, 26, 27 and the impact of time pressures on the research process.19, 21, 22, 23, 24, 26, 27 Other less frequently reported barriers included logistical hurdles, such as meeting disability accommodations,17 as well as challenges with retention of patient partners in studies where patient partners experienced changing life circumstances or disease recurrence, limiting their ability to continue their participation in the project.19, 26 In some instances, partnerships were found to burden both patients and researchers with emotional tolls and burnout. For example, in one study patient partners experienced an emotional burden if confronted with the possibility of a recurrence of their disease while participating in patient partner activities.19 With respect to additional challenges, two studies cited financial resource shortage issues,21, 26 one study referred to fearing patient ‘tokenism’,17 and one study indicated group conflict21 as key barriers to forging and maintaining partnerships.

25Studies also noted several facilitators to engaging in patient partnerships. One common facilitator cited was valuing the patient partner role,17, 22, 23, 29 which was accomplished when parties including the research leadership team, team members and patient partners themselves expressed the value and worth of patient partner contributions. Aligned with this facilitator, it was also identified that clear role descriptions and responsibilities for patient partners were important factors for facilitating effective partnerships.25, 26, 27 For patient partners, this often meant the use of explicit policies or guiding documentation to support these descriptions.25, 26, 27 Additionally, studies reported that meeting the personal needs of patient partners (ie disability accommodations, scheduling adjustments, provision of refreshments and transportation) helped to remove barriers to partnerships.17, 20, 21, 25, 26 Other studies noted the critical role that compensation for time and work played in facilitating patient partnerships.25, 26, 30 An atmosphere of camaraderie between researchers and patient partners, as well as among patient partners themselves,21, 23 was also reported as facilitative of patient partnership. Additional facilitators noted were purposeful recruitment of patient partners through existing organizations,22 sufficient time and space for partnership,18, 24, 27 and flexibility and responsivity (eg personal contact23 and preparedness27) of the research team.23, 26, 27

26In included studies, patient partnership was predominantly evaluated by the research team itself. In four included studies, researchers themselves evaluated the impact of partnership,20, 21, 24, 26 and in seven studies, this was jointly evaluated with patient partners or other stakeholders.19, 22, 23, 27, 28, 29, 30 Only two of the included studies used an external, independent review process.17, 25 As this field continues to develop, there may be further opportunity to look to external and independent evaluation of partnership on both outcomes and process. This may reduce the risk for bias, as well as enhance those facilitators related to team cohesion, trust and roles/responsibilities.

27Building on the work of previous systematic reviews focused on patient engagement, this scoping review sought to synthesize current available evidence surrounding patient partnership in the research process and to identify impact on research process and research outcomes. Our findings draw attention to the paucity of research where patient partnership is evaluated quantitatively, as all studies included in this scoping review drew on qualitative techniques, with interviews and focus groups primarily used to evaluate partnership strategies. Across studies, teams grappled with the concrete impact of their partnership strategies. Results highlighted that patient partners took on various roles within the research process, and experienced personal impact related to knowledge and skill development, relationship building, and contribution of the ‘patient voice’ to research. Other researchers have noted similar findings in relation to patient engagement research in general (ie not solely focused on patient partners), whereby there is an evaluative focus on the process of patient engagement and its personal impact on patients.31, 32

28There remains pressure for researchers to conceptualize concrete outcomes of patient partnerships to determine the ‘value add’ of involving patients in health research.12 Examples of attempts to quantify the benefits of engaging patient partners in research include the development and application of economic equations to theoretically quantify the financial value of engagement,33 and the development and testing of measures such as the Patient Engagement in Research Scale (PEIRS)34 and Public and Patient Engagement and Evaluation Tool (PPEET),35 among others.36 The evidence base derived from use of these tools is developing and remains in the early stages. Moreover, evaluation of impact of patient partnership has tended to focus on impact on the research process versus impact on the outcomes of the research.36

29Quantifying the impact of patient partnerships on research outcomes remains elusive. As such, where patient partnership seems best evaluated may be when it is positioned as a quality indicator around research processes. Patient partnerships represent complex relationships within research teams, which may be enacted through a wide variety of strategies, dependent on the needs of the project, resources available, and the capacity and desire for involvement of patient partners themselves. Manafo, Petermann, Vandall‐Walker and Mason‐Lai7 describe varying levels of patient engagement that progress from ‘learning and informing’ to ‘leading and supporting’. Building off of this framework, findings from this scoping review support the need for enhanced guidance and understanding regarding how and what aspects of patient partnership can and should be evaluated. Examples of aspects of patient partnership evaluated in included studies include patient partner training sessions,18 patient experiences of research participation,19, 25 and personal impacts of participation experienced by patient partners.20, 21, 23 Within the extant body of literature on patient engagement, other examples of process and outcome measures for effective engagement exist that could reasonably be extended to evaluation strategies for patient partnership (see Esmail et al31).

30Existing tools, such as those found within the Centre of Excellence on Partnership with Patients and the Public (CEPPP),37 may be of use in the evaluation process. Examples of evaluation strategies for assessing the quality of patient partnerships include use of initial, midpoint and end‐of‐project surveys to understand the evolving relationships between patient partners and researchers, and what behaviours support productive partnerships.38 Researchers wishing to report the impact of patient partner involvement in their work may consider doing so using a standardized method such as the GRIPP2 reporting checklist39 for reporting patient and public involvement in research. The GRIPP2 reporting checklist prompts researchers to report the specific definition of patient and public involvement within the study, the methods and stages at which they were involved, and outcomes and impact of involvement, among other factors.39 Standardization and transparent reporting of patient partner involvement in research is urgently needed in this field, where ambiguity and divergence in definitions of patient partnership, methods for partner involvement, and reporting results related to patient partner involvement remain the norm. By positioning patient partnerships as a research orientation, the focus of patient‐engaged research would be to include the patient voice in all stages of research, in order to facilitate the many benefits of patient partnerships to both health research and patient partners themselves. As others have articulated, there may also be a ‘moral obligation’ to include those impacted by research in its development.31

31In our review, we found that a number of research teams were thoughtful about enfranchising patient partners in the research process. When working with patients as partners in a research team, power dynamics can influence the effectiveness and successfulness of partnership.40 Consenting patients to be patient partners positions patients as ‘subjects that contribute’ rather than as equal team members. By omitting the consenting process in partnership, power balances are more evenly distributed between researchers and patients, recognizing that each individual contributes unique skills and expertise to meet research goals. However, omitting consent processes with patient partners did not always serve to completely ameliorate uneven power dynamics in research teams, and several included studies in this review reported that power imbalances remained problematic barriers within their projects. Some literature suggests that unbalanced power dynamics may be mitigated through co‐production of all processes and deliverables between researcher and partner, capitalizing on all assets, expertise, and capabilities to ensure equal and reciprocal partnerships.41, 42 Researcher and patient partner co‐production can assist to avoid tokenism and power imbalances.43

32Our search strategy targeted patients who were not consented as study participants as we aimed to ensure that patient partners were represented as fully integrated team members and not research participants who were then called upon to take on ancillary roles. The process of integrating patient partners into the research study team has been utilized and reported in various ways throughout the literature, such as patient partners contributing to research as members of ‘discussion groups’ – whereby patient partner contributions help to steer long‐ or short‐term research decisions, but are not treated as research data44; or co‐design team members – whereby ‘users’ and ‘producers’ of health services interventions work in concert to design individual services or interventions.45 Arguments for removing the consent process from patient partners participating as members of the study team include a departure from paternalist ideals of consenting patient partners to studies ‘owned’ by researchers,45 and shifting the lens of ‘protection’ of patients to a mutually beneficial partnership between those with lived experience and those researching that experience.45

33While being enfranchised as a research team member clearly has merits, this approach is not without risk. Removing informed consent processes in order to equalize power imbalances has the potential to introduce an element of risk to patient partners, if done without adequate preparation to facilitate informed partnerships. Inadequate preparation for partnership inhibits engagement by both failing to provide the prerequisite background and skills for patients to fully participate and by introducing risk to patient partners via the potential for misinterpretation or misinformation regarding roles and expectations for the partnership. Patient partners who are not adequately informed may approach their roles seeking direct benefits from their participation, particularly in instances where patients seek expertise from researchers or clinicians for personal reasons.

34Resource constraints are known to be another barrier to implementing effective partnership strategies.1, 31, 32 Inherently, compensation, or lack thereof, can contribute to power imbalance between researchers and patient partners. When addressing equity among a team, consideration should be made to address whether and how patient partners are to be compensated for the skills and expertise they bring forward. At present, challenges exist regarding how to quantify the worth of lived experience, and what is to be provided on volunteer basis.46 However, several resources exist on the topic of remuneration of patient partners in health research, such as guidelines from the CIHR47 and PCORI,48 as well as public policies such as those from Diabetes Action Canada.49 Generally, available guidelines suggest remuneration in scope with the level of involvement of patient partners within the study. To attenuate power imbalances when working with patient partners, we suggest having open conversations with patient partners to identify whether monetary compensation is expected and what can feasibility be provided to ensure all participating parties – researchers and patients alike – feel valued for the experiences and knowledge which they contribute.

35Finally, our scoping review revealed one of the most common barriers across studies to be a lack of training for patient partners. To resolve this issue, it is our contention that partnership requires thoughtful preparation of both researchers and patients to create an informed patient partner engagement experience. Aspects of preparation include clear education, role definition and guidance for both researchers and patient partners,50 which may serve to maximize patient partner involvement and contributions to research outcomes, rather than limiting patient partners to only providing reflections of their personal experiences.2 Manafo et al51 identified common barriers to the practice of engagement across the spectrum of research activities, including inadequate preparation of both patients and researchers for engagement. As the Canadian Health Research Roadmap II suggests, transformational research involves the integration of stakeholders, including patients, into the research process.8

36Brett et al11 reported that a lack of preparation for patient partners led to unease and misunderstandings on the part of patient partners as to their roles and expectations. Confusion, conflict and disappointment in not being given medical support for their conditions through research partnerships have also been reported.11 In light of this, researchers may want to consider including an explicit discussion with patients prior to partnership to be clear about the fact that their health‐care needs are separate from research involvement, per se. To support informed patient partnership, we endorse the use of formal recruitment and training programmes for patient partners, and the inclusion of guidelines for patient partnership such as those available from the Canadian Stroke Prevention Network's Patient Engagement Resources.52 In addition, the Canadian Institutes of Health Research Working Group on Ethics in Patient Engagement in Research has developed a draft document that provides guidance on useful considerations for researchers wanting to engage patient partners in research.40 Research teams who are purposeful about patient partner roles, compensation and expectations may be best positioned to mitigate barriers and implement patient partner engagement.

37The literature included in this review largely reflects studies involving adults with chronic, long‐term health conditions as patient partners in research. To this end, only one included study involved youth as patient partners in their work. Thus, findings may not be applicable to researchers seeking to undertake patient partnerships with youth, or outside of the area of chronic conditions.

38Secondly, only studies that did not consent patient partners were included in this review. While this team acknowledges that in some instances, partnership and participant roles may overlap, our interest was specifically in those studies evaluating the impact of patient partnership when patient partners were part of the research team ‘only’, and not holding dual roles as a research partner and participant. As such, the considerable patient engagement and patient partnership literature that required patient partners to formally consent to be part of the research team was excluded. While it is likely that this literature would offer further insights regarding impact of patient partnerships, that would be a different focus than the present review.

39Patient partnership in research represents an opportunity to leverage the knowledge, skills and experience of not only researchers and clinicians, but also those that research directly impacts – patients and families – to collaborate towards creating impactful and meaningful change in how research is conducted. This scoping review has synthesized the available literature on patient partnership with respect to contributions, as well as barriers and facilitators to partnership. Consideration of the implementation of patient partner preparation strategies to create informed partnerships may help to mitigate unbalanced power dynamics. Finally, the operationalization of patient partnership roles varies across study designs. Guidance is needed for researchers in terms of evaluating patient partnership processes and outcomes according to the level of patient engagement.

40The authors declare that there is no conflict of interest.