Document characteristics
After screening and quality appraisal of 2633 information sources, a final sample of 72 articles describing 62 studies informed the review [several studies had multiple articles [2, 46,47,48,49,50,51,52,53,54,55,56,57,58,59,60,61,62,63,64,65,66,67,68,69,70,71,72,73,74,75,76,77,78,79,80,81,82,83,84,85,86,87,88,89,90,91,92,93,94,95,96,97,98,99,100,101,102,103,104,105,106,107,108,109,110,111,112,113,114,115, 119]; Additional file 1: Appendices D, E]. Twenty-two studies were from the United States (35.5%), 20 were from the United Kingdom (32.3%), 14 from Canada (22.6%), three from Australia (4.8%), and three involved international collaborations (4.8%). The studies were a mix of descriptive (52 studies, 83.9%) and interventional (10 studies, 16.1%) with diverse outcomes [Additional file 1: Appendices D, E].
Analysis and synthesis
A grand total of 287 explanatory accounts were developed from the 62 studies. From these, 26 context-mechanism-outcome (CMO) configurations were identified and synthesized into an initial program theory. After review, feedback, and refinement by stakeholders, the revised program theory consisted of seven contexts and eight mechanisms [Additional file 1: Appendices F, G]. Five mechanisms supported the hypotheses put forth by the preliminary program theory.
Mechanisms were predominantly associated with sets of contexts. Interactions between contexts and the mechanisms “deciding to become involved in patient-oriented research [M1],” “recognizing valuable experiential knowledge [M2],” “cultural competence [M3],” “reducing power differentials [M4],” “respectful team environment [M5],” “supporting patient partners to feel valued [M6],” and “readiness to research [M7]” resulted in the intermediate outcome of a “sense of trust [iO].” Trust then acted as a mechanism [M8] which triggered the final outcome, “empowered patient-centred lens [FO]”. The following sections describe the set of eight mechanisms and their respective triggering contexts. Embedded resources that enabled each mechanism were identified. The relationships between mechanisms and the outcomes are presented below. Individual hypotheses that were supported from the preliminary program theory are identified.
Deciding to become involved in patient-oriented research [M1]
Twenty-five articles revealed individuals are motivated to make a decision to become members of patient-oriented teams for various reasons [2, 46,47,48, 59, 65, 67, 68, 73, 74, 79, 81,82,83, 92, 93, 95, 97, 100, 107,108,109,110,111, 119]. Individually-centred motivations (potentially self-benefitting), and consistent with the first hypothesis [H1] of the preliminary program theory, altruistically-centred (potentially benefitting others) were identified for both patient partners and academic researchers. There was partial support of the third hypothesis [H3] in the preliminary program theory as both positive and negatively perceived health care experiences could motivate an individual to become a patient partner.
For patient partners, individually-centred motivations were the desire to learn new skills [68, 73, 110, 111] and to support the individual to reframe their self-identity away from being a sick person [73, 110, 111]. While honoraria did not appear to motivate individuals to participate in patient-oriented research, patient partner stakeholders of the review team further clarified that an unintended consequence of offering honoraria is that academic researchers may mistakenly perceive members of the public are predominantly drawn to patient-oriented research solely because of these funds.
Altruistically-centred motivations for patient partners included the desire to show gratitude and to ‘pay back’ their health care system for the good care they had received [107, 110, 111], desire to improve the living conditions of one’s community [74, 81, 92, 100, 107], and to improve the care of future patients [2, 47, 48, 59, 65, 73, 79, 82, 83, 93, 95, 97, 107, 110, 111]. Moreover, individuals can decide to become a patient partner after being a participant in a research study [46,47,48, 59, 108, 109, 119].
For academic researchers, individually-centred motivations were new opportunities for funding and for career advancement [73]. Altruistically-centred motivations included the potential for new knowledge creation, better understanding of patients’ perspectives, and improved patient care [73, 114].
Four articles identified disincentives for participating in patient-oriented research. Individuals who anticipated the work associated with being a patient partner or academic researcher would compete for time required in other aspects of their life, such as time required for family responsibilities, clinical duties, teaching, were less likely to participate in patient-oriented research [46, 67, 73, 107].
Recognizing valuable, experiential knowledge [M2]
Active, meaningful engagement of a patient partner(s) within a research study assumes that an academic researcher(s) has made two decisions: the patient partner(s) possesses experiential knowledge that is relevant to the study, and it is anticipated that this knowledge can be utilized by the team in a manner that will improve that research study.
Nineteen articles revealed contexts that appeared to trigger these decisions for academic researchers [46,47,48, 53, 55, 57, 59, 70, 81, 84, 92, 93, 96, 100, 102, 108, 109, 112, 113]. These contexts included that the patient partner was a highly-regarded member of a group or community [47, 48, 53, 81, 84, 93, 100, 102, 112], the patient partner had lived within a rural or urban community for many years [47, 48, 59, 84, 92, 96, 113], the patient partner was a member of a recognized patient group or healthcare-related charity with a history of conducting research [55, 57, 70], the patient partner was a former study participant of the academic researcher [46,47,48, 59, 108, 109, 119].
Developing cultural competency [M3]
Fourteen articles indicated the importance of cultural competency [46,47,48, 53, 66, 69, 73, 74, 79, 81, 87, 93, 95, 106]. Academic researchers who collaborate with vulnerable individuals and communities, but know little about the people, culture, and setting of their research, can face difficult interactions with groups and community members [53, 73, 74, 81, 87, 93]. For example, individuals who have endured discrimination and historical mistreatment can experience fear, anger, and/or be suspicious of researchers’ intentions in reaction to their presence within their community [73, 74, 87, 93]. Resources that supported academic researchers’ development of cultural understanding included their completion of cultural competency training [74, 87, 106], and their participation in a guided tour of the community early on during a study’s planning. During the tour, academic researchers listened to first-hand accounts from residents and community leaders about how their history and environment had negatively affected them [87]. Resources that supported patient partners to feel more comfortable included having academic researchers’ formally acknowledge to community leaders at the onset of study planning that historical mistreatment underlies community’s mistrust of their presence [87], having balanced representation of patient partners and academic researchers on the research team [74, 79, 87], rotating team meetings between community-based and academic-based locations [69] and providing informal opportunities for patient partners and academic researchers to try to get to know each such as sharing a meal prior to the start of a team meeting or taking time after a meeting ends to socialize with one another [46,47,48, 66, 74, 87, 95].
Reducing power differentials [M4]
Twenty-three articles focussed on supporting groups and communities facing social inequities [46,47,48, 53, 65, 66, 68, 69, 72,73,74, 79, 81, 84, 87, 89, 93, 95, 97, 100, 102, 112, 114] Citizens of these groups and communities have endured historical discrimination and mistreatment, lived in impoverished communities and under-serviced areas, and/or are living with a stigmatized medical condition [47, 48, 53, 65, 68, 73, 81, 84, 87, 93, 97, 100, 102, 112, 114].
Consistent with the second and sixth hypothesis [H2, H6] of the preliminary program, a resource that afforded some power back to vulnerable groups and communities was the establishment of an advisory council which represented their collective interests during the study. Highly-regarded individuals within these groups and communities such as Indigenous tribal leaders, tribal elders, traditional healers, clergy, parents, and a rural mayor held membership on advisory councils [47, 48, 53, 81, 84, 93, 100, 102, 112]. Advisory committees made decisions throughout the course of a study granting of permission for outsiders to enter their community, providing insight to academic researchers regarding their community’s structure, and ensuring that activities introduced as part of the study were culturally-appropriate for their members. Several advisory councils also prepared a formal memorandum, or agreement, of understanding [47, 48, 72, 81, 100, 112]. This resource supported collective clarification of the roles, responsibilities, and deliverables that were required of the advisory council, patient partners, academic researchers and any other parties that were to be involved in the research study. A team may refer back to its memorandum of agreement if any questions or concerns arise [72].
Beyond the establishment of advisory councils, other resources were used to reduce perceived differentials in power between team members. These included having equal numbers of patient partners and academic researchers on the research team [74, 79, 87], rotating team meetings between community-based and academic-based locations [69] and requiring that academic researchers attend each team meeting [73]. Two sets of field notes, one recorded by patient partners and one recorded by academic researchers were included as official records of team meetings [89]. Informal activities such as sharing a meal before a team meeting afforded opportunities for team members to try to get to know each other [46,47,48, 66, 74, 87, 95].
Respectful team environment [M5]
Consistent with the fourth hypothesis [H4] of the preliminary theory, ten articles indicated academic researchers used resources to cultivate, and maintain, team environments that were perceived by patient partners as being respectful and safe to participate in [2, 46, 63, 70, 73, 80, 97, 108, 109, 111]. Examples included scheduling research team meetings at times that were convenient for patient partners to attend [46, 73, 80], formally delegating protected time during team meetings where a patient partner(s) could speak to whatever topic they saw fit [108, 109], and recognizing that a patient partner’s medical condition can require them, in the moment, to have additional time to express their thoughts [80, 97]. Academic researchers also made efforts to explain medical jargon and avoid using acronyms as much as possible when interacting with their patient partners [46, 70]. Feelings of vulnerability can occur for patient partners who have endured discrimination and historical mistreatment, are living with a stigmatized medical condition, and/or are experiencing deteriorated health [2, 63, 110, 111]. During these moments, it was important patient partners were provided with emotional support from team members.
Supporting patient partners to feel valued [M6]
Ten articles supported the hypothesis that patient partners are vigilant of cues from academic researchers that appeared to signal whether those academics valued them as authentic team members [66, 67, 74, 79, 80, 82,83,84, 108, 109]. Travelling to a patient partner’s home in order to meet [80], formally delegating protected time during research team meetings where a patient partner(s) was free to talk about topic(s) they perceived as being important [108, 109], and providing patient partners with individual honoraria and/or covering their travel and childcare costs [66, 67, 79, 84, 108, 109] were actions made by academic researchers that demonstrated to patient partners that their input was valued. A community-based organization could also be recognized with an honorarium [74].
The value of patient partners’ expertise was also memorialized on study-related brochures and other text-based information sources. These information sources identified patient partners as official members of a care team [82, 83], announced ongoing achievements made by members of a community during a study [81] and repeatedly emphasized that patient partner expertise enabled the study to thrive [97]. Patient partners were also formally identified as a co-investigator or co-author of the study [66, 108, 109].
Readiness to research [M7]
The preliminary program theory did not take into account patient partners’ preparedness for patient-oriented research. Four articles indicated that when moments of tension were reported between academic researchers and patient partners, these appeared to be triggered by academic researchers’ perceptions regarding patient partners’ lack of methodological knowledge and/or inexperience with processes required for the study ([73, 85, 87, 119]; Additional file 1: Appendix C).
Twenty articles revealed that educational background and previous experience with patient-oriented research were contexts that appeared to signal, to academic researchers, that an individual was either ‘research ready’ or would likely require only minimal training to become ready to research [53, 55, 57, 60, 62, 63, 68,69,70, 73, 76, 82,83,84,85, 87, 90, 96, 97, 106]. Educational backgrounds signaled a patient partner was research ready if the individual had previously completed training in a reputable, patient-oriented research program [76], the individual was a member of a recognized patient group or healthcare-related charity with a history of conducting research [55, 57, 70]. Individuals with a medical degree or medical background were also perceived as being research ready [97]. Academic researchers provided training during the course of the study in order to prepare patient partners without research backgrounds [53, 60, 62, 63, 68, 69, 73, 82,83,84, 87, 90, 96, 106]. The review team’s patient partner stakeholders further advocated the advancement of patient partner expertise requires that individuals have ongoing opportunities for collaboration with academic researchers in patient-oriented research.
Developing a sense of trust [iO1/M8] and empowering a patient-oriented lens [FO]
Fifteen studies revealed that the development of a sense of trust was important for maintaining cooperative relationships between patient partners and academic researchers [47, 48, 66, 67, 72, 74, 79, 84, 87, 95, 102, 106, 108, 109, 119]. Resources used by academic researchers to develop patients’ sense of trust included providing honoraria to patient partners [66, 67, 79, 84, 108, 109], seeking the consensus of all team members during decision-making [106], incorporating patient partner input into the study [108, 109], making visible efforts to try to get to know members of the community where a study was set [47, 48], and being willing to engage in open, frank discussion with community members about their intentions in the community [74].
While the preliminary program theory emphasized empowerment of patient partners [H3, H8], it did not overtly position a role for trust. The review team’s patient partner stakeholders recognized, and emphasized, this was an important omission because just as patients, families, and caregivers need to perceive a sense of trust during any interaction with their health care system, trust also lies at the centre of the patient partner-academic researcher relationship. These stakeholders cautioned that if patient partners do not develop a sense of trust with academic researchers, a patient-oriented research study will not achieve its full potential. This is because members of the public, including experienced patient partners, will not be empowered to feel confident to fully open up to researchers regarding their lived health journeys, how they perceive their personal health experiences can be of benefit to the research study, and make suggestions and decisions about a research study.