Patient partners’ perspectives of meaningful engagement in synthesis reviews: A patient‐oriented rapid review

Abstract Background A growing literature describes promising practices for patient‐oriented research (POR) generally; however, those for systematic reviews are largely derived through the lens of a researcher. This rapid review sought to understand meaningful engagement in synthesis reviews from the patient partner (PP) perspective. Design The review team comprised PPs, librarians, SCPOR staff and academic faculty. We searched OVID MEDLINE and EMBASE, ProQuest Nursing and Allied Health, and core POR websites. Documents describing PP reflections on their involvement in synthesis reviews were included. Screening and data extraction were conducted by two independent reviewers. Thematic analysis was employed to identify themes in the data regarding PP perceptions of engagement in synthesis reviews. Results The literature search yielded 1386 citations. Eight journal articles and one blog post were included. Seven studies focused on conducting systematic reviews on a particular health or patient‐related topic to which PP involvement was an important part and two studies focused specifically on the experience of including PP in synthesis reviews. PPs engaged in the review process through a variety of mechanisms, levels and stages of the review process. Three major themes emerged from the data: (1) foster partnerships through team development, (2) provide opportunities for outcomes valued by PP and (3) strengthen the research endeavour. Conclusion Fostering partnerships through team development is foundational for meaningful engagement in synthesis reviews. It requires sensitively balancing of various needs (eg overburdening with contributions). Meaningful involvement in reviews has both personal and research benefits. Patient Involvement Patient partners were equal collaborators in all aspects of the review.


| INTRODUC TI ON
A new approach to health-care research in Canada was initiated in 2011 when the Canadian Institutes of Health Research (CIHR) launched the Strategy for Patient Oriented Research (SPOR). The intent was 'to demonstrably improve health outcomes and enhance patients' health care experience through integration of evidence at all levels in the health care system'. 1 Patients were to become coinvestigators within health-care research, and their voices were to be regarded within a research context formerly dominated by academics and clinicians. In this way, Patient-Oriented Research (POR) democratizes health research and enables stakeholders, especially patients, to shape the health care system and empowers individuals and communities with the opportunity to influence change. 2 A patient, according to CIHR, is an overarching term that includes individuals with personal experience of a health issue and informal caregivers, including family and friends. 3 POR is defined as 'a continuum of research that engages patients as partners, focuses on patient identified priorities and improves patient outcomes. This research, conducted by multidisciplinary teams in partnership with relevant stakeholders, aims to apply the knowledge generated to improve health care systems and practices'. 3 Patient partner (PP) involvement in research has been promoted as a means to ensure the relevance of research agendas and questions, facilitate the recruitment of study participants, improve the likelihood of funding success and enhance the dissemination of results. [4][5][6][7][8] Engagement of PPs in research has proposed value to patients, to researchers and to the improvement of the research process, all of which may ultimately impact health care, policies and outcomes. 2 Impacts to PPs include increased knowledge of the research process; a sense of empowerment and fulfilment; and the increased ability for patients to advocate for themselves. 2,4,9 Researchers who engage in POR benefit from gaining a richer understanding of a health condition or disease from the patient's lived experience and an expansion of research opportunities and scope. 2,10 The operationalization of POR requires further attention as engaging patients in health-care research is a new team dynamic and tokenism can be a pitfall. 11 Integrated knowledge translation insists that having all stakeholders and knowledge users represented within POR teams ensures a collaborative team from the outset; one in which all members are equal and one in which all stakeholders are vested in the findings. 12 Resulting reported improvements to the research process include the identification of research priorities that are relevant to patient needs, more appropriate 'real world' research questions and designs, increased credibility and applicability of findings, and improved quality of knowledge translation materials. 2,4,10 Similarly, a recent scoping review 13 identified five primary PP roles in research: steering committee membership; advisory committee membership; consultation; co-design of knowledge translation; and participation in research tasks. Specific examples of steering committee member roles included providing input on design of instruments and tools used in the study, 14 while advisory roles included assisting with systematic review 15 and conference planning. 16 PP consulting activities involved sharing the ways in which intervention would affect patients, 17 and specific research activities which PPs readily participated in encompassed development of research questions, interview guides, research priorities, data analysis and attendance at study briefings. [18][19][20] The overarching aims of POR approaches are to optimize health outcomes and quality of life in ways that are important to patients, which ultimately relies on the extent to which patients are meaningfully engaged in the research process. 21 Existing frameworks 22,23 describe recommendations to foster meaningful patient engagement in research. Pollock and colleagues 24,25 focused on methods of involving stakeholders in a specific kind of research, namely systematic reviews. Our review extends this work by investigating how research teams can ensure that PP contributions to synthesis reviews (SRs) are meaningful from the PP's perspective.

| The objective of this study is to better understand best practices in engaging patients in synthesis reviews
Our team synthesized published and grey literature on patients perspectives of meaningful engagement in SRs, employing Hamilton et al's definition: '…the planned, supported and valued involvement of patients in the research process within an interactive team and positive research environment that facilitates effective contributions by patients or their surrogates to help to produce important outcomes while benefiting the patients or their surrogates'. 23 We aimed to answer two questions: 1. What are the characteristics of a review team that create an 'interactive team and positive research environment that is conducive to effective contributions by patients'?
2. How can research teams conduct their reviews to ensure 'planned, supported and valued involvement of patients'?

| Team composition and training
The review team comprised two PPs (AME, TP), two stakeholders representing the Saskatchewan Centre for Patient-Oriented K E Y W O R D S consumer participation, knowledge synthesis, knowledge translation, patient and public involvement, patient engagement, patient-oriented research, systematic reviews Research (SCPOR) (CS, KD), two librarian faculty (AG, CB) and two research faculty (BB, DG). PPs were recruited from a Saskatchewan Centre for Patient-Oriented Research (SCPOR) patient partner pool.
Both PPs were very familiar with the health-care system although they had never participated in a POR rapid review. An introduction to rapid review methods was provided in the first meeting which included a simulation activity based on identifying chocolate chip cookie recipes within cooking literature. A description of the activity is provided in Supporting information.
All team members were fully involved in all aspects of the review from determining the question to dissemination. Training was integrated into team meetings, including pre-readings and instructional activities (eg mock data extraction examples), and undertaken by the entire team. Additional informal training was provided as requested. Meetings prior to mid-March 2020 were held in-person. In mid-March 2020, all non-essential workers in the team's location were required to work remotely due to the COVID-19 pandemic. All subsequent meetings were held online.
The transition to virtual meetings occurred as we finished data extraction. In Table 1, we describe in more detail how PP were involved in the conduct of the review, and team reflections on their involvement.

| Study selection
We included documents which described or reflected on PP perspectives of meaningful engagement in SRs. PPs had to have participated in the conduct of a synthesis review, but there was no restriction on the level of engagement. The document had to focus on meaningfulness from the patient's perspective. Thus, we included only reflective articles and grey literature (eg reports) written by or in collaboration with patients documenting their experiences on SRs. Documents describing patient engagement in SRs where patients were participants in the research and did not contribute to the planning, conduct or dissemination, were excluded. SRs include systematic reviews, scoping reviews, realist reviews, meta-syntheses, meta-analyses, qualitative reviews, health technology assessments and clinical practice guidelines. Patient engagement in other kinds of reviews (eg narrative reviews) or research was excluded. We were interested in methodologically rigorous reviews which are more typically employed to guide research agendas (eg scoping reviews) or practice/policy (eg systematic reviews, realist reviews) specifically.
All members of the team contributed equally to the screening process. Screening was conducted by pairs of two independent reviewers using a screening form developed and piloted a priori. First, the titles and abstracts of citations were reviewed. Then, the full text of citations deemed of possible interest was retrieved and reviewed.
Disagreements were resolved through consensus at both stages.
Covidence software was employed to facilitate the screening process.

| Data extraction
We employed Hamilton et al's definition 23 of meaningful patient engagement as the framework for extracting data about PP engagement in the review process. Data were extracted by pairs of independent reviewers using an extraction form that was developed and piloted a priori (Supporting information). All members of the team contributed equally to extracting the following data: • Descriptive data about the publication (ie authors, publication year, journal, country of corresponding author, country in which study was conducted, sponsorship and funding); • Descriptive data about the study (ie overall aim and purpose of the study, total number of authors, number of PP authors, study design, synthesis review team composition, PP engagement and training); • Qualitative data regarding the mechanisms for PP engagement and training, and PP perceptions of that engagement. Excerpts from the study pertaining to PPs involvement in a systematic review were coded for voice (patient or research team voice).

| Data synthesis
Frequencies were calculated for descriptive data about the publication and the study. Qualitative data were analysed using thematic analysis as described by Braun and Clarke. 27 Initially, all team members independently coded the data to become fully familiar with the data set. This initial coding ensured deep understanding of the dataset by each team member but yielded eight unique coding schemes.
The extracted data by the POR team were then coded and themed

| Study selection
The database searches yielded 1386 citations (1373 from databases, 13 from the grey literature search). After removing 297 duplicates, 1089 citations were screened. Forty articles were excluded upon review of the full text for the following reasons: lack of reflection on PP experiences contributing to a synthesis review (n = 20); PPs were not involved in a synthesis review (n = 17); and the protocol had insufficient information to determine inclusion status (n = 3). Five studies and one companion study were removed during data extraction due to insufficient data from patient perspective. Eight journal articles and one blog were included ( Figure 1). TA B L E 1 Guidance for reporting involvement of patients and the public (GRIPP2) checklist. 37

Aim
To collaboratively involve patients as research partners at all stages in the rapid systematic review research project to ensure PP perspective in the review process and outcomes.

Methods
PPs were recruited through a post on the SCPOR website for research opportunities. Training was integrated into team meetings throughout the review, and additional informal training was provided as needed.
PPs were included as collaborators on the research team, participating fully in all aspects of the review from research question refinement to knowledge translation which had equal input for the duration of the project.

Study results
Through their active contributions, PPs helped craft the research question and provided input and insights at each stage of the review. PPs encouraged a team culture of deliberately considering multiple viewpoints during team discussions. PPs lead some stages of the review, specifically the knowledge translation plan and the GRIPP2 checklist content.

Discussion and conclusions
PPs contributed important PP perspectives and lay language. PPs asked provocative and necessary questions at each step of the process and often provided an invaluable lens not only to the research process but to the content as well.
Reflections/critical perspective Patient Partners: • PPs may want to choose a topic of interest as it is challenging to review content without a personal interest.
• PPs received positive feedback from other team members that validated PP contribution during discussions.
• PPs were able to ask questions and for clarification during and after meetings.
• PPs gained valuable experience in health research and would get involved in future rapid reviews.
• PP learned about other team members expertise in their areas of work and perspectives and appreciated a 'Wonderful opportunity to build a multidisciplinary team that can learn something from everyone'.. • PP had opportunities to step outside of their comfort zone by participating in the review, including being accepted to present at an international conference.
• PP should be involved in rapid reviews. PP noted some negatives of their involvement: • Project required extended timelines due to COVID-19.
• Additional and on-going support may be required if a PP has limited or no experience with rapid reviews or research process. Other team members': A lack of clarity could have impeded participation without PP's willingness to ask questions and to communicate needs and workload. Timelines were significantly affected by the pandemic. The willingness of PP to continue with this project in very trying circumstances beyond the agreed time was a significant factor in the completion of this project.

| Characteristics of included studies
Studies employed the following methods: questionnaires/survey reflections from PPs that were part of the engagement activities, but direct patient quotes were not used. One study used exclusively the researchers' voices to describe events and experiences of the PPs and their participation in the review. 32 In the remaining studies, it was less clear as to who was lending their voice to the interpretation of events. One study 33 did not include any PPs as authors, so the voice portrayed was likely that of the researchers'. Two studies 3435 also appear to be presented in the voice of the researchers. However, one 28 had PP authors, so it is possible they lent much to the interpretation and summary. Characteristics of the nine included studies are provided in Table 2.
Only two of the included studies focused specifically on the experience of including PPs in systematic reviews (including one metasynthesis). 28,30 Seven of nine studies were reviews conducted on a particular health or patient-related topic to which PP involvement was an important part. 15,29,[31][32][33][34][35] The latter included reflections by the PPs or the research team on engagement in the review process.
Research teams provided formal and informal training in synthesis review methods to PPs, which occurred at specific points in the review or, in one case, interspersed through the project.
Four studies mention training specific to synthesis review methods. 15,30,32,33 Three studies provided some form of training but it was unclear if supports were specific to SR methodology: monthly training sessions, 31 written instructions 28 and informal training and support. 29 Two studies were unclear or did not report on training for PPs. 34

| Synthesis
Three major interrelated themes emerged from thematic analysis: 1) foster partnerships through team development; 2) provide opportunities for outcomes valued by PP; and 3) strengthen the research endeavour. Below we describe the themes and subthemes. The relationship of the themes and subthemes to our two research questions is provided in 'Reflection from one of the contributors (WP), however, highlighted that although this was an interesting meeting and they were pleased to help, because we were unable to give them clear guidance on which interventions they should be using (or not using), they felt that the meeting had limited benefit for them'.

| Strengthen the Research Endeavour
PP and other researchers recognized that engaging PP meaningfully in a systematic review influenced research priorities and enhanced research relevance and quality.

Influence research priorities
PPs provided an added perspective to the research process by addressing different priorities for researchers. Once PPs became engaged in the synthesis review, their exposure to the literature provided them the opportunity to see the important gaps in health research, which they were able to identify. Challenges to PP engagement in SRs are documented in Table 4.

| Study limitations
The COVID-19 pandemic forced the team to pivot to a fully online mode of communication. This occurred while we were doing data extraction, but mostly impacted thematic analysis. The team acknowledged that reduced team member availability and online meeting format had the potential to significantly affect our timelines. We chose to engage CHASR to expedite the first stages of analysis. This did not adversely affect analysis quality but was a change from our planned methods.
Pollock et al 24 commented on the limited reporting of PP involvement in systematic reviews, a concern we echo. We were often left wondering whose voice was being represented-the PP's, the In this review, we included a variety of studies and document types as the literature exploring PP perspectives was limited. A consequence of this choice was the use of secondary data in our analysis. In-depth qualitative data analysis was not as strong as it would be with primary data sources. Thus, we have chosen not to develop a model of PP perspectives on meaningful engagement, but rather provide some initial recommendations to guide researchers until a more robust literature is available (Table 5).

| CON CLUS ION
Meaningful PP involvement in SRs can benefit the PP and research.
Fostering partnerships by taking the time for developing the team is foundational for meaningful engagement in synthesis reviews. For involvement to be meaningful for PPs, not only do team dynamics and processes need to be conducive, but PPs need to see benefits, personal or as impacts on research, from their work. There must be a sensitive balancing of various needs (eg overburdening with contributions). While this rapid review has provided preliminary guidance on how and what review teams can do, more high-quality research about the PP perspective is needed to inform a robust model of meaningful engagement in SRs from the PP perspective.

ACK N OWLED G EM ENTS
We would like to acknowledge the contribution of the Canadian Hub for Applied and Social Research (formerly the Social Science Research Laboratories) at the University of Saskatchewan for data analysis support.

CO N FLI C T O F I NTE R E S T
All authors declare that they have no conflict of interest.

AUTH O R CO NTR I B UTI O N S
CB, AEM, TP, BB, KD, AG, DG and CS (all authors) made substantial contributions to conception and design, acquisition of data, analysis and interpretation of data of this review. All authors contributed to drafting the manuscript and gave final approval of the manuscript.

DATA AVA I L A B I L I T Y S TAT E M E N T
Data sharing is not applicable to this article as no new data were created.

G R E Y LITE R ATU R E S E A RCH
A three-staged grey literature search was undertaken in January-February 2020, with the initial step being the identification of patient orientated research websites, likely to have relevant information on patients' perspective of meaningful engagement in SRs. An initial list of core grey literature sources was distributed to the research team and feedback was sought based on the team's expertise and experience in this area. Additional grey literature sources were identified at this stage. As we were employing a rapid review methodology, the intent of the grey literature search was to be systematic but not exhaustive; this was similar to the approach with the database search strategy.
The second stage was a preliminary review of all the grey literature sources identified in the previous stage. This process involved a review of the website and its content to determine if a deeper inves-