Clarifying the mechanisms and resources that enable the reciprocal involvement of seldom heard groups in health and social care research: A collaborative rapid realist review process

Abstract Objective Public and patient involvement is increasingly embedded as a core activity in research funding calls and best practice guidelines. However, there is recognition of the challenges that prevail to achieve genuine and equitable forms of engagement. Our objective was to identify the mechanisms and resources that enable the reciprocal involvement of seldom heard groups in health and social care research. Methods A rapid realist review of the literature that included: (a) a systematic search of CINAHL, PsycINFO, PubMed and Open Grey (2007‐2017); (b) documents provided by expert panel members of relevant journals and grey literature. Six reference panels were undertaken with homeless, women's, transgender, disability and Traveller and Roma organizations to capture local insights. Data were extracted into a theory‐based grid linking context to behaviour change policy categories. Main results From the review, 20 documents were identified and combined with the reference panel summaries. The expert panel reached consensus about 33 programme theories. These relate to environmental and social planning (7); service provision (6); guidelines (4); fiscal measures (6); communication and marketing (4); and regulation and legislation (6). Conclusions While there is growing evidence of the merits of undertaking PPI, this rarely extends to the meaningful involvement of seldom heard groups. The 33 programme theories agreed by the expert panel point to a variety of mechanisms and resources that need to be considered. Many of the programme theories identified point to the need for a radical shift in current practice to enable the reciprocal involvement of seldom heard groups.


| INTRODUC TI ON
While there is no consensus on one definition for public and patient involvement (PPI) nor in relation to the terminology used (e.g engagement and involvement are often used interchangeably or with different connotations), there is a growing abundance of academic and grey literature on the merits, impact and experiences of PPI in health and social care research. [1][2][3][4][5] In the UK, the National Institute for Health Research (NIHR) has set up the platform INVOLVE to promote and share best practices for PPI. In Canada, the Institute for Health Research (CIHR) has developed a strategy for Patient Outcome Research (POR). While in Ireland, the Health Research Board (HRB) launched the PPI Ignite Awards in 2017 which are focused on enabling institutional-wide PPI responses within universities. As PPI becomes more embedded as a core activity in many national and international funding calls, the evaluative literature has shifted to capture impacts. [6][7][8] A recent systematic review and modified Delphi process to capture an agreement on the principles underpinning PPI point to a new focus on what is required to sustain and embed these principles within university structures. 9 More recently, Palmer et al 10 have provided a welcome depth to the theory on the processes of co-production and co-design within mental health improvement and system redesign. These shifts are to be welcomed but caution and recognition of the challenges are also prevalent in the recent literature. [11][12][13][14][15] In particular, there is a recognition that PPI partners' involvement can be tokenistic often at the lower level of consultation. 1 Ocloo and Matthews outline a range of reasons why achieving genuine patient involvement presents challenges, citing in particular a lack of diversity of those becoming involved. They call for the inclusion of more diverse populations via the implementation of more inclusive and democratic models of engagement that are embedded in co-design. 11 The merits of overcoming the often-identified challenges of engaging diverse voice or "seldom heard" groups have been stressed where different perspectives informed by, for example, socioeconomic status, ethnicity, health status or gender can provide deeper insights in designing and implementing a trial. 16 There is also a recognition in the literature of the challenges of engaging diverse populations. These engagements often occur at the lowest levels focused on consulting rather than involving. 11,12 The demands on researchers to involve more diverse populations and to move to higher levels of co-produced involvement bring new demands to support, develop and sustain. Key to this is to understand and map efforts, initiatives and strategies designed to enhance the collaborative capacity skills of researchers, the public and those working within the health system. [14][15][16] The focus of this paper was to identify the strategies that may help overcome the often-identified challenges of engaging seldom heard groups. Being seldom heard means that existing structures and processes in organizations including universities and health and social care providers may not be adequately matched to the needs of all members of the public. The aim for this rapid realist review (RRR) was to clarify the mechanisms and resources required to enable seldom heard people to be involved in health and social care research. A characterization of seldom heard has been provided in a protocol paper published prior to the review. 17

| Study methods
An RRR approach was chosen as it explicitly allows for the engagement of knowledge users throughout the review process. 17 In contrast to a systematic review, the RRR builds an understanding of why and how things work (programme theories). A detailed rationale for and characterization of the review process has been outlined in our protocol paper. 18 The RRR adhered to the RAMESES realist publication standards guides with adaptations to streamline and accelerate the process as advised in the literature. 19,20

| Establishment of an expert panel
An expert panel convened in March 2018 consisting of members who have experience in health and social care systems, PPI, co-design, emancipatory research and people and organizations representing provision (6); guidelines (4); fiscal measures (6); communication and marketing (4); and regulation and legislation (6).

Conclusions:
While there is growing evidence of the merits of undertaking PPI, this rarely extends to the meaningful involvement of seldom heard groups. The 33 programme theories agreed by the expert panel point to a variety of mechanisms and resources that need to be considered. Many of the programme theories identified point to the need for a radical shift in current practice to enable the reciprocal involvement of seldom heard groups.

K E Y W O R D S
behaviour change wheel, co-design, health and social care research, public and patient involvement, rapid realist review, seldom heard seldom heard groups (Appendix S1). All expert panel members are co-authors of this paper. The first meeting clarified the scope and the overarching RRR question as being "What are the mechanisms that enable the reciprocal involvement of seldom heard groups?" Key terms were defined and agreed by the group (Appendix S2). The search strategy, conditions and participants were reviewed (Table 1), and the inclusion and exclusion criteria were developed.
The expert panel created an extraction template (Appendix S3) to ensure that mechanisms and resources would be captured. The expert panel's initial discussion focused on the importance of developing policy responses that could be implemented from the review within their respective organizations. It was therefore agreed that the template would extract contexts linked to adopted policy categories as noted in the behaviour change wheel (BCW). 21 The linking to the BCW policy categories was used in the template to examine the developed programme theories by providing a contextual overview. The BCW is a recent but increasingly popular taxonomy to assist the development and implementation of behaviour change interventions. 22 In this review, the BCW was used to describe the mechanisms and related resources of involving seldom heard groups in relation to specific contextual factors.

| Reference panel process
Reference panels are local sounding boards undertaken in an RRR to ensure that the review and the developed programme theories are inclusive to the experience of those "on the ground." 18

| Nature of data set
After screening and comparison with inclusion/exclusion criteria (Table 2) 1. Systematic reviews: Three systematic reviews including an Australian-based review on barriers to the participation of socio-economically disadvantaged groups in health research and strategies on how to increase engagement. 23 The second was a UK-based review focused on black and minority ethnic group-PPI involvement in health and social care research. 24 A UKbased evidence synthesis was the third review concentrated on health and social interventions for inclusion health for people with experiences of homelessness, drug use, imprisonment and sex work. 25 2. Empirical articles: Eight in total including a UK study presenting how seldom heard groups and social care services establish inclusive involvement practice. 26 The second was a Canadian article on engaging frail older people and caregivers in research and decision making. 27 The third was a US study outlining what worked in engaging diverse non-English speaking communities. 28 The fourth was an Irish paper on undertaking participatory learning and action approach in developing GP communication guidelines with migrants. 29  amongst African American women. 30 The sixth was an Australian study on how a randomized controlled trial developed an inclusive methodological approach co-designed by people affected by severe mental illness. 31 The seventh was a regional Australian study on engaging families in the design of child protection measures. 32 The eight study was from the UK that explored differences be- Irish-based report on what was learned from an emancipatory research partnership involving community and university partners to capture the experiences of Travellers and people with experience of the asylum process in respect to access of public services. 40 The second was a UK summary of a seminar exploring best practice themes around service user involvement for women facing multiple disadvantages. 41 The third was an Irish report to capture the process of people in recovery being involved in identifying and conducting their own research using a peer lead approach. 42 A summary of findings about the extracted articles and reference panels linked to mechanisms and resources is outlined (Appendix S6).

| Agreed programme theories linked with behaviour change wheel policy categories
The expert panel reconvened in July 2018 and reviewed the extracted data. The expert panel via consensuses validated and prioritized 33 programme theories linking them to the adopted contextual policy categories as noted in the BCW. 22 The programme theories were generated from the review and synthesis of findings from the literature, refinement in discussions with reference panels and via the final consensus meeting with the expert panel ( Figure 2).
Below is a summary overview of the mechanisms of 33 programme theories (statements on what works) that emerged linked to the six BCW policy categories (Boxes 1-6).
The review found that engagement with seldom heard groups needs to occur in safe, accessible and inclusive spaces. 25,32,33,[38][39][40][41][42] Ensuring spaces are accessible needs to be guaranteed by undertaking an audit at the start of the research, which should be monitored and reviewed throughout the project. 25,[40][41][42]  The majority of papers and reference panels stressed that early engagement with community partners was a key enabler to shape the research process from the outset. 25

Secondary Exclusion Criteria
• Studies without descriptions of any intervention or mechanism.
• Studies that do not report any outcome or result.
• Studies without health and social research elements.
• Unable to obtain further information to make assessment.

Inclusion Criteria
• Both quantitative and qualitative studies.
• Both published and grey literature (e.g websites, reports, dissertations and theses).

Duplicates removed (n = 70)
Full-text articles assessed for eligibility using string 2 (n = 266) Being present with community partners and ensuring feedback is ongoing as agreed with community partners were identified in the reference panels. Ensuring engagement and outputs are flexible and innovative emerged in most of the literature and in the reference panels. [23][24][25][26][27]31,[35][36][37][38][39][40][41][42] The importance of all partners creating and reviewing a co-design checklist emerged in the Adshead and Dubula study. 37 They outlined tensions that occurred during the study. This happened between academics who were working towards project timelines agreed by the grant funders and the desire of community partners, who wished to advance emancipatory work at their own pace. 37 Developing guidelines/protocols were identified as a key mechanism to support diverse involvement, ensuring flexibility and clarity on data ownership and usage. 23,36 The reference panels with Pavee Point and TENI identified the need to make funding available to include psychological supports as required. The importance of funding for alternative outputs as identified by community partners such as accessible lay summaries was noted in five studies. 24,25,28,34,37 Flexibility in payments to co-researchers emerged in the literature and in the reference panels. 23,25,28,34,40 Dublin Simon Community and DFI reference panels noted that peer researchers could often be in receipt of social welfare payments and research payments could have an impact on this. As such, vouchers, cash or whichever is most appropriate for the community partners were suggested as alternatives. The Kaiser study identified the importance of allocating funding to celebrate success with community partners. 34 Including funding to cover the cost of involvement emerged as a mechanism in most of the literature. [23][24][25][26][27][28][29][30][31][32][33][34]36,[38][39][40][41] DFI stressed that this was key as often it was expected that they as an organization should cover these costs.
Providing resources for the development of a research partnership emerged significantly in Robinson et al, 32 who also emphasized the need to be present to develop trust.
Identifying what all partners would like to achieve from the study should be prioritized. 37 The Nguyen et al 28  Two studies identified that university ethics applications and guidelines should be reviewed to always assume competence of study participants and to enable processes to seek verbal consent on an ongoing basis. 34,38 The time it takes to develop research partnerships should be included in funding calls and specific calls should be focused on codesign. This was the case in two studies where funding was made available to specifically to undertake emancipatory and participatory action learning approaches. 37,42 Ensuring flexibility in funding schemes to enable community partners to identify needed supports was identified in the three systematic reviews. [23][24][25] Finally, the inclusion of community partners in the development and evaluation of funding calls was identified within the reference panel process. The Longford Women's Link also stressed that training for community partners on evaluations be made available to support their capacity to be involved in the process.  26,28,[30][31][32][33][34][35][36][37][38][39][40][41][42] The review notes structural challenges that need to be navigated such as ethics, payments and access to university resources for community partners and sustainable funding to enable participation. 23,25 Having multiple partners working on a project often results in tensions given the remits of different agenda that can emerge. 37,38 It is important that time and adequate flexible resources are made available to celebrate success and achievements. 32 The review also found that funders have a key role to play to enable the reciprocal involvement of seldom heard groups. 29,40 As the shift away from a "fund and forget model" continues, the need to resource pre-engagement and long-term partnerships grows stressed in the reference panels as crucial to enable involvement (Appendix 6). We would urge that further contributions be made to the literature on how reciprocal projects with seldom heard groups have resulted in reforms and changes linked to the six BCW policy categories. Additional work should also expand and refine these programme theories by engaging with other seldom heard groups.

| D ISCUSS I ON AND CON CLUS I ON S
We recognize that there are limitations within this work in particular that an RRR is not a comprehensive search, review and synthesis of the literature. However, the methodological strength and process of engagement allowed for a broad engagement with seldom heard people and organizations representing them. The RRR process enabled us to capture local expertise via our six reference panels and the insights they shared captured valuable mechanisms that enhanced the richness of the review. RRRs can support PPI initiatives by producing programme theories of what works. This work contributes to a field where there has been little evidence of what works. It is evident from our developed programme theories that mechanisms and associated resources need to combine and interact to enable and sustain the reciprocal involvement of seldom heard groups in health and social care research.

ACK N OWLED G EM ENTS
We would like to thank those who participated in the reference pan-

CO N FLI C T O F I NTE R E S T
The authors declare that they have no competing interests.

AUTH O R S' CO NTR I B UTI O N S
All the authors have made significant intellectual or practical contributions towards the development of the RRR. ÉNS drafted this paper, and all authors read, edited and approved the final manuscript.