The purpose of a paper in the social sciences designed around a case study is to thoroughly investigate a subject of analysis in order to reveal a new understanding about the research problem and, in so doing, contributing new knowledge to what is already known from previous studies. In general, the structure of a case study research paper is not all that different from a standard college-level research paper. However, there are subtle differences you should be aware of. Here are the key elements to organizing and writing a case study research paper.
As with any research paper, your introduction should serve as a roadmap for your readers to ascertain the scope and purpose of your study. The introduction to a case study research paper, however, should not only describe the research problem and its significance, but you should also succinctly describe why the case is being used and how it relates to addressing the problem. The two elements should be linked. With this in mind, a good introduction answers these four questions:
- What was I studying? Describe the research problem and describe the subject of analysis you have chosen to address the problem. Explain how they are linked and what elements of the case will help to expand knowledge and understanding about the problem.
- Why was this topic important to investigate? Describe the significance of the research problem and state why a case study design and the subject of analysis that the paper is designed around is appropriate in addressing the problem.
- What did we know about this topic before I did this study? Provide background that helps lead the reader into the more in-depth literature review to follow. If applicable, summarize prior case study research applied to the research problem and why it fails to adequately address the research problem. Describe why your case will be useful. If no prior case studies have been used to address the research problem, explain why you have selected this subject of analysis.
- How will this study advance new knowledge or new ways of understanding? Explain why your case study will be suitable in helping to expand knowledge and understanding about the research problem.
Each of these questions should be addressed in no more than a few paragraphs. Exceptions to this can be when you are addressing a complex research problem or subject of analysis that requires more in-depth background information.
II. Literature Review
The literature review for a case study research paper is generally structured the same as it is for any college-level research paper. The difference, however, is that the literature review is focused on providing background information and enabling historical interpretation of the subject of analysis in relation to the research problem the case is intended to address. This includes synthesizing studies that help to:
- Place relevant works in the context of their contribution to understanding the case study being investigated. This would include summarizing studies that have used a similar subject of analysis to investigate the research problem. If there is literature using the same or a very similar case to study, you need to explain why duplicating past research is important [e.g., conditions have changed; prior studies were conducted long ago, etc.].
- Describe the relationship each work has to the others under consideration that informs the reader why this case is applicable. Your literature review should include a description of any works that support using the case to study the research problem and the underlying research questions.
- Identify new ways to interpret prior research using the case study. If applicable, review any research that has examined the research problem using a different research design. Explain how your case study design may reveal new knowledge or a new perspective or that can redirect research in an important new direction.
- Resolve conflicts amongst seemingly contradictory previous studies. This refers to synthesizing any literature that points to unresolved issues of concern about the research problem and describing how the subject of analysis that forms the case study can help resolve these existing contradictions.
- Point the way in fulfilling a need for additional research. Your review should examine any literature that lays a foundation for understanding why your case study design and the subject of analysis around which you have designed your study may reveal a new way of approaching the research problem or offer a perspective that points to the need for additional research.
- Expose any gaps that exist in the literature that the case study could help to fill. Summarize any literature that not only shows how your subject of analysis contributes to understanding the research problem, but how your case contributes to a new way of understanding the problem that prior research has failed to do.
- Locate your own research within the context of existing literature [very important!]. Collectively, your literature review should always place your case study within the larger domain of prior research about the problem. The overarching purpose of reviewing pertinent literature in a case study paper is to demonstrate that you have thoroughly identified and synthesized prior studies in the context of explaining the relevance of the case in addressing the research problem.
In this section, you explain why you selected a particular subject of analysis to study and the strategy you used to identify and ultimately decide that your case was appropriate in addressing the research problem. The way you describe the methods used varies depending on the type of subject of analysis that frames your case study.
If your subject of analysis is an incident or event. In the social and behavioral sciences, the event or incident that represents the case to be studied is usually bounded by time and place, with a clear beginning and end and with an identifiable location or position relative to its surroundings. The subject of analysis can be of a rare or critical event or focus on a typical or regular event. The purpose of studying a rare event is to illuminate new ways of thinking about the broader research problem or to test a hypothesis. Critical incident case studies must describe the method by which you identified the event and explain the process by which you determined the validity of this case to inform broader perspectives about the research problem or to reveal new findings. However, the event does not have to be a rare or uniquely significant to support new thinking about the research problem or to challenge an existing hypothesis. For example, Walo, Bull, and Breen conducted a case study to identify and evaluate the direct and indirect economic benefits and costs of a local sports event in the City of Lismore, New South Wales, Australia. The purpose of their study was to provide new insights from measuring the impact of a typical local sports event that prior studies could not measure well because they focused on large "mega-events." Whether the event is rare or not, the methods section should include an explanation of the following characteristics of the event: when did it take place; what were the underlying circumstances leading to the event; what were the consequences of the event
If your subject of analysis is a person. Explain why you selected this particular individual to be studied and describe what experience he or she has had that provides an opportunity to advance new understandings about the research problem. Mention any background about this person which might help the reader understand the significance of his/her experiences that make them worthy of study. This includes describing the relationships this person has had with other people, institutions, and/or events that support using him or her as the subject for a case study research paper. It is particularly important to differentiate the person as the subject of analysis from others and to succinctly explain how the person relates to examining the research problem.
If your subject of analysis is a place. In general, a case study that investigates a place suggests a subject of analysis that is unique or special in some way and that this uniqueness can be used to build new understanding or knowledge about the research problem. A case study of a place must not only describe its various attributes relevant to the research problem [e.g., physical, social, cultural, economic, political, etc.], but you must state the method by which you determined that this place will illuminate new understandings about the research problem. It is also important to articulate why a particular place as the case for study is being used if similar places also exist [i.e., if you are studying patterns of homeless encampments of veterans in open spaces, why study Echo Park in Los Angeles rather than Griffith Park?] and, if applicable, what type of human activity involving this place makes it a good choice to study [prior research reveals Echo Park has more homeless veterans].
If your subject of analysis is a phenomenon. A phenomenon refers to a fact, occurrence, or circumstance that can be studied or observed but with the cause or explanation to be in question. In this sense, a phenomenon that forms your subject of analysis can encompass anything that can be observed or presumed to exist but is not fully understood. In the social and behavioral sciences, the case usually focuses on human interaction within a complex physical, social, economic, cultural, or political system. For example, the phenomenon could be the observation that many vehicles used by ISIS fighters are small trucks with English language advertisements on them. The research problem could be that ISIS fighters are difficult to combat because they are highly mobile. The research questions could be how and by what means are these vehicles used by ISIS being supplied to the militants and how might supply lines to these vehicles be cut? How might knowing the suppliers of these trucks from overseas reveal larger networks of collaborators and financial support? A case study of a phenomenon most often encompasses an in-depth analysis of a cause and effect that is grounded in an interactive relationship between people and their environment in some way.
NOTE: Evidence that supports the method by which you identified and chose your subject of analysis should be linked to the findings from the literature review. Be sure to cite any prior studies that helped you determine that the case you chose was appropriate for investigating the research problem.
The main elements of your discussion section are generally the same as any research paper, but centered around interpreting and drawing conclusions about the key findings from your case study. Note that a general social sciences research paper may contain a separate section to report findings. However, in a paper designed around a case study, it is more common to combine a description of the findings with the discussion about their implications. The objectives of your discussion section should include the following:
Reiterate the Research Problem/State the Major Findings
Briefly reiterate the research problem you are investigating and explain why the subject of analysis around which you designed the case study were used. You should then describe the findings revealed from your study of the case using direct, declarative, and succinct proclamation of the study results. Highlight any findings that were unexpected or especially profound.
Explain the Meaning of the Findings and Why They are Important
Systematically explain the meaning of your case study findings and why you believe they are important. Begin this part of the section by repeating what you consider to be your most important or surprising finding first, then systematically review each finding. Be sure to thoroughly extrapolate what your analysis of the case can tell the reader about situations or conditions beyond the actual case that was studied while, at the same time, being careful not to misconstrue or conflate a finding that undermines the external validity of your conclusions.
Relate the Findings to Similar Studies
No study in the social sciences is so novel or possesses such a restricted focus that it has absolutely no relation to previously published research. The discussion section should relate your case study results to those found in other studies, particularly if questions raised from prior studies served as the motivation for choosing your subject of analysis. This is important because comparing and contrasting the findings of other studies helps to support the overall importance of your results and it highlights how and in what ways your case study design and the subject of analysis differs from prior research about the topic.
Consider Alternative Explanations of the Findings
It is important to remember that the purpose of social science research is to discover and not to prove. When writing the discussion section, you should carefully consider all possible explanations for the case study results, rather than just those that fit your hypothesis or prior assumptions and biases. Be alert to what the in-depth analysis of the case may reveal about the research problem, including offering a contrarian perspective to what scholars have stated in prior research.
Acknowledge the Study's Limitations
You can state the study's limitations in the conclusion section of your paper but describing the limitations of your subject of analysis in the discussion section provides an opportunity to identify the limitations and explain why they are not significant. This part of the discussion section should also note any unanswered questions or issues your case study could not address. More detailed information about how to document any limitations to your research can be found here.
Suggest Areas for Further Research
Although your case study may offer important insights about the research problem, there are likely additional questions related to the problem that remain unanswered or findings that unexpectedly revealed themselves as a result of your in-depth analysis of the case. Be sure that the recommendations for further research are linked to the research problem and that you explain why your recommendations are valid in other contexts and based on the original assumptions of your study.
As with any research paper, you should summarize your conclusion in clear, simple language; emphasize how the findings from your case study differs from or supports prior research and why. Do not simply reiterate the discussion section. Provide a synthesis of key findings presented in the paper to show how these converge to address the research problem. If you haven't already done so in the discussion section, be sure to document the limitations of your case study and needs for further research.
The function of your paper's conclusion is to: 1) restate the main argument supported by the findings from the analysis of your case; 2) clearly state the context, background, and necessity of pursuing the research problem using a case study design in relation to an issue, controversy, or a gap found from reviewing the literature; and, 3) provide a place for you to persuasively and succinctly restate the significance of your research problem, given that the reader has now been presented with in-depth information about the topic.
Consider the following points to help ensure your conclusion is appropriate:
- If the argument or purpose of your paper is complex, you may need to summarize these points for your reader.
- If prior to your conclusion, you have not yet explained the significance of your findings or if you are proceeding inductively, use the conclusion of your paper to describe your main points and explain their significance.
- Move from a detailed to a general level of consideration of the case study's findings that returns the topic to the context provided by the introduction or within a new context that emerges from your case study findings.
Note that, depending on the discipline you are writing in and your professor's preferences, the concluding paragraph may contain your final reflections on the evidence presented applied to practice or on the essay's central research problem. However, the nature of being introspective about the subject of analysis you have investigated will depend on whether you are explicitly asked to express your observations in this way.
Problems to Avoid
One of the goals of a case study is to lay a foundation for understanding broader trends and issues applied to similar circumstances. However, be careful when drawing conclusions from your case study. They must be evidence-based and grounded in the results of the study; otherwise, it is merely speculation. Looking at a prior example, it would be incorrect to state that a factor in improving girls access to education in Azerbaijan and the policy implications this may have for improving access in other Muslim nations is due to girls access to social media if there is no documentary evidence from your case study to indicate this. There may be anecdotal evidence that retention rates were better for girls who were on social media, but this observation would only point to the need for further research and would not be a definitive finding if this was not a part of your original research agenda.
Failure to Document Limitations
No case is going to reveal all that needs to be understood about a research problem. Therefore, just as you have to clearly state the limitations of a general research study, you must describe the specific limitations inherent in the subject of analysis. For example, the case of studying how women conceptualize the need for water conservation in a village in Uganda could have limited application in other cultural contexts or in areas where fresh water from rivers or lakes is plentiful and, therefore, conservation is understood differently than preserving access to a scarce resource.
Failure to Extrapolate All Possible Implications
Just as you don't want to over-generalize from your case study findings, you also have to be thorough in the consideration of all possible outcomes or recommendations derived from your findings. If you do not, your reader may question the validity of your entire analysis, particularly if you failed to document an obvious outcome from your case study research. For example, in the case of studying the accident at the railroad crossing to evaluate where and what types of warning signals should be located, you failed to take into consideration speed limit signage as well as warning signals. When designing your case study, be sure you have thoroughly addressed all aspects of the problem and do not leave gaps in your analysis.
Case Studies. Writing@CSU. Colorado State University; Gerring, John. Case Study Research: Principles and Practices. New York: Cambridge University Press, 2007; Merriam, Sharan B. Qualitative Research and Case Study Applications in Education. Rev. ed. San Francisco, CA: Jossey-Bass, 1998; Mills, Albert J., Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research. Thousand Oaks, CA: SAGE Publications, 2010; Putney, LeAnn Grogan. "Case Study." In Encyclopedia of Research Design, Neil J. Salkind, editor. (Thousand Oaks, CA: SAGE Publications, 2010), pp. 116-120; Simons, Helen. Case Study Research in Practice. London: SAGE Publications, 2009; Kratochwill, Thomas R. and Joel R. Levin, editors.Single-Case Research Design and Analysis: New Development for Psychology and Education. Hilldsale, NJ: Lawrence Erlbaum Associates, 1992; Swanborn, Peter G. Case Study Research: What, Why and How? London : SAGE, 2010; Yin, Robert K. Case Study Research: Design and Methods. 6th edition. Los Angeles, CA, SAGE Publications, 2014; Walo, Maree, Adrian Bull, and Helen Breen. “Achieving Economic Benefits at Local Events: A Case Study of a Local Sports Event.” Festival Management and Event Tourism 4 (1996): 95-106.
In this chapter we present the findings from the 22 case studies. We provide a rationale for why NPT was found to be the most comprehensive and predictive programme theory for PPI and present the 16 NPT informed mechanisms that were tested within the case studies. We then summarise the key findings and list the contexts that enabled mechanisms. CMO tables are provided, illustrating salient actions within the case studies, potential contexts, plausible mechanisms and potential outcomes. Organised under the major case study actions, data are presented to illustrate how the CMO tables were arrived at. We draw on specific case studies, including their NPT radar plots, to provide illustrative exemplars. The chapter concludes with evidence of PPI impact and outcomes.
Normalisation Process Theory as the candidate programme theory being tested
Our testing within case studies was framed by the 16 NPT mechanisms grouped under the four headings (see Chapter 3) and presented in Table 11. When applied to the 22 case studies, NPT121,134 had sufficient explanatory power to predict which case studies had PPI embedded as normal practice; therefore, it predicted both policy outcomes and the processes and infrastructure required for effective PPI that had positive outcomes both methodologically and morally. Using NPT as a MRT developed out of health service research, we directly interpreted PPI as the complex intervention, which then shaped our data collection tools and analytical framework (see Chapter 3).
Summary of key findings
This is one of the first studies to contribute significantly to our understanding of PPI as a concept, through the analysis of a large number of case studies, selected to capture varying levels of PPI embeddedness, in order to understand how it may change over time during the research process.
In testing whether or not topic area had an influence on the embedding of PPI, we found similarity between the nature of the clinician–patient relationship and the way PPI was enacted. This was further influenced by the topic area’s history of PPI and the tacit models that had evolved. For example, we found PPI in IDD studies to be framed by an empowerment model and resonant of the long history of partnership working between practitioners and service users. Well-established PPI structures in some topic areas such as dementia provided a pragmatic source for the lay perspective, but we found unintended outcomes for this model, including the outsourcing of PPI to external parties with little researcher engagement. It also exemplified a ‘one-off’ model whereby PPI was limited to a single task.
Researcher perceptions of what lay representatives could be expected to understand or be interested in, coupled with a lack of effort to communicate in an accessible way, sometimes led to limiting lay involvement in parts of the research process, potentially limiting impact. However, we also found in areas such as basic science and tissue banks, where PPI has traditionally been seen as having a limited role, that research teams were enabling PPI, leading to improved relevance of research priorities and questions.
While still challenging for a number of studies and funders, recruiting a diverse range of lay people was enhanced when the study setting interfaced with a diverse population. Particular PPI structures helped, including an outreach model with either a specific lay person or a co-ordinator acting as a bridge to a broader community. The quality of such connections was important to create the context for involvement. The resulting impact was culturally sensitive, participant-focused research designs.
In contrast, researchers’ descriptions of ‘usual suspects’ as lay representatives reflected a perception of concerns about the professionalisation of PPI. However, our findings suggested this was at odds with reality and with the advantages of the experienced lay representative being able to develop a number of skills that helped their role, and enabling a career trajectory for the lay person within PPI.
An important finding in both the survey and the case studies was the dual role, whereby the lay advisor was also a study participant. Although this had the benefit of real-time feedback about the impact of the research design and enhanced a sense of ownership of the study for lay respondents, there was also evidence of conflicts of interest and a blurring of roles. In some case studies the dual role was a deliberate PPI design; however, in others it clearly demonstrated that involvement was not understood as being different from participation, and highlighted the level of PPI drift away from INVOLVE definitions. It was also indicative of a lack of training or experience in PPI, particularly in novice researchers. Training or on-the-job experience not only helped researchers to differentiate between involvement and participation, but also enhanced an understanding of what both parties bought to the table. Development of positive relationships underpinned by trust was the minimal requirement for any form of PPI; this led to improved self-worth for lay people, and increased researchers’ confidence in their work. Breakdown in this relationship had a profound effect on levels of PPI embeddedness over time.
It was clear from the case studies that PPI was most likely to be embedded when hosted in well-resourced research centres with a dedicated budget and PPI co-ordinator. PPI groups and panels had continued engagement with the research host organisation, were reimbursed equitably and had social interaction with the research teams; these are crucial to the sustaining of positive relationships. Resourcing PPI in such a way also enabled a fully entwined model of PPI, in which lay people and researchers worked seamlessly together, with research priorities and research questions created by the lay people involved. This generated some research programmes that would not have been run without lay involvement. However, embedding PPI to this level required not only resources but also time to develop.
We found that funder requirements played a key role in influencing levels of reimbursement. In addition, how PPI was operationalised in individual studies could be traced back to the funder’s ethos of PPI. Within funding organisations there was a growing interest in evaluating processes and outcomes of PPI within studies. However, even with funder expectations, only a limited number of case studies were reflecting on and evaluating PPI in an ongoing way to improve the quality of PPI processes and outcomes.
The findings suggested there were eight enabling contexts for the mechanisms (Box 5). Five of these (funder, topic area and design, resources, organisation of PPI and research host) were expected, based on the literature, and shaped our sampling framework to ensure a range of case studies as a test-bed. The other three (training, positive relationships and positive experience) became visible during data collection and analysis.
Enabling contexts C1 Funder requirements for PPI in research.
As described in Chapter 3, through an iterative process we tested candidate CMO configurations within each case study and discussed our findings with case study respondents in the tracking interviews, with our reference groups and finally at the four regional events. We identified six CMO configurations that explained salient case study actions, potential contexts, plausible mechanisms and potential outcomes, both intended and unintended. CMO configurations focus on immediate outcomes within a study. It was not possible to include longer-term outcomes of a study, as these evolve over a longer period of time. Tables of each CMO are presented and then illustrated by data and case study exemplars. Within exemplars, NPT radar plots will be shown, and any change in PPI embeddedness over time will be illustrated through tracking radar plots.
Purpose, structure and role of patient and public involvement
Table 12 illustrates the CMO configuration for this action. Funder requirements were a significant enabling context, and had resulted in a more structured approach to PPI. It was also facilitated by host organisations with an established PPI infrastructure, such as an established group or panel, and access to training. At the time of data collection, this enabling context was provided by some topic areas (dementia and diabetes mellitus) with network support for PPI. Having adequate resources to maintain this group in terms of administration and reimbursement was vital for sustained engagement with PPI in research.
Context, mechanism and outcome configuration: purpose, role and structure for PPI
For some case studies, the purpose, structure and role for PPI were an inherent part of the research design. For example, CS03 (see Box 10) and CS23 (see Box 21) were funded to coproduce an intervention (self-management website and mobile phone applications for young people with diabetes mellitus). Other case studies sometimes had to craft a PPI structure from new because of the funder’s requirements.
Case study 03: diabetes mellitus Experts by experience, different levels:
Case study 23: diabetes mellitus Four YPD as advisory group.
Researchers were particularly aware that when applying for specific grants there were even stronger imperatives to have PPI as a funding requirement. PPI was recognised as a part of the contemporary research landscape, and some reflected that having formalised PPI as a requirement was beneficial, as experience of good PPI usually leads to more in the future:
more researchers are willing to engage, whether that’s because they want to or whether because they have to because of the grant application, I think there’s a bit of both there, but then again, that’s fine because those that . . . engage because they have to kind of start to begin to see the worth of it anyway.
Overall, the funder respondents clearly asserted that PPI both in research and at funder and system level had developed significantly over the previous 5 years, describing a journey along a continuum of learning and understanding of PPI, despite noting that there was still some way to go until it was fully embedded as normal practice:
I think organisationally that’s been a sea-change . . . Externally with researchers I sense there has broadly been a change but there’s still a lot to do, but certainly the quality of the PPI anecdotally that we’re getting in our applications is on the up, possibly to do with . . . everything being reviewed by a public contributor.
Structures and processes for PPI within funder organisations themselves inevitably varied and strongly reflected the influence of their core funding source, mission, scale and length of service. PPI structures had developed either quite systematically or more recently and organically as part of the overall PPI movement.
Working closely with INVOLVE, two overarching programme management organisations and two research programmes that are funded by central government had, over a number of years, developed similar, embedded structures for PPI within their organisations. However, the sheer volume of applications to process could potentially result in the PPI representatives having to work with a conveyor belt approach. The industrial scale of PPI as metaphor was also picked up by one of the lay representatives:
I think, having started from a low base, it’s [PPI] now in danger I think of almost becoming an industry; that is that everybody concerned with health care has to have some public involvement, and that goes for GP practices, hospitals, and very much in research, all areas of research . . . It sometimes strikes me that it’s a kind of bandwagon. Don’t get me wrong, I think it’s one of the most useful things in the health economy, but it seems to have made jobs for an awful lot of people, let’s put it that way.
The other funding bodies taking part in the RAPPORT study had only recently started to implement PPI strategies in their organisations. A government-funded research council had endorsed its PPI strategy only a year before and there was now a strategic vision around PPI. The structure of this funding organisation was less complex and was led by a member of staff responsible for both public involvement and engagement. This organisation had a PPI panel of around 20 members who were offered the opportunity to be involved in particular funded projects; alternatively, the organisation would ask specific medical charities to nominate lay representatives for particular condition-specific projects. Another large national funder, financed through endowments, had a different stance on PPI, focusing more on public engagement:
patient and public involvement doesn’t just have to be people in a focus group or a patient representative on a steering committee, it can, you can use kind of more engagement approaches that can still inform and can kind of be part of the research process . . . sometimes when I talk to people in the patient and public involvement sphere there’s quite a narrow definition of how they see patient and public involvement, and so yeah, I can see it a bit wider than that.
Public engagement was seen as a key policy driver within this organisation, and strategically it focused on facilitating research teams to improve its public engagement, with PPI being seen as operationalised at research team level.
We also interviewed two representatives from condition-specific medical research charities. Their PPI strategies had gradually developed over recent years:
we’re currently looking at expanding it, so it is something that’s definitely on the radar of people within the charity, it’s just the technicalities. I think, you know, everyone’s definitely willing to do it and I know that our Medical Director is very keen on PPI, and I think it is almost evolving organically . . .
A major finding from the interviews with funder representatives was that their stances or values (and thus PPI structures and processes) were actively shaped by the origin of their funding and their histories. The funding organisations that received government public funding as part of a national research agenda clearly articulated the reasons for PPI in methodological terms:
It [PPI], you know, is likely to improve recruitment. It makes sure that it meets patient needs so that when it gets out the other end if you like into the implementation phase that it can actually have a possibility of having impact on an individual with a said condition.
In contrast, the medical charities reliant on fundraising spoke of PPI in terms of an ethical rationale:
we get our money from the public, from people with the condition, so it’s really important that we include viewpoints of people with the condition in our decision.
Hence, funders influenced PPI in subtly different ways depending on their organisational values. Case studies funded by NIHR funding streams were required to have PPI as a different way of working within the actual research process because of the emphasis on the methodological importance of PPI. In contrast, those funded by charities tended to have less focus on a requirement for PPI in the actual research process, with structures in their organisation concentrated more on ensuring PPI in funding decisions.
Structures for patient and public involvement
Host organisations, and in particular CLAHRCs, were instrumental in providing a PPI infrastructure. This often involved having a designated lead for PPI. Six of the case studies had a dedicated PPI co-ordinator who was separate from the research team, and a further case study had a research team member who took on the role of the PPI co-ordinator as part of their duties. Case study 21 (Box 6) had a colocated co-ordinator who, as a non-researcher, was seen to bridge the gap between researchers and the public:
Case study 21: IDDs The research centre hosts a large PPI group made up of parents.
she’s become a great interface between us and the families because she helps, she’s . . . she knows enough about research to be able to explain what we do, but equally doesn’t know enough that she can challenge me or others in the team to explain more clearly what we’re asking of families. And I think that’s probably the greatest reason for our success has been the sort of creation of that role and the development of it.
Case study 21 exemplified fully embedded PPI which was enabled by all eight identified contexts. It also demonstrated an ‘entwined’ model of PPI in which research and PPI were wrapped together much like a double helix. It was hosted within a research centre primarily funded by a charity (C1) with a topic area (children with severe cognitive disabilities) (C3) where active involvement of parents was seen as a prerequisite of meaningful research. The study design (systematic review) (C3), compared with the ongoing qualitative research being undertaken in the programme of work, required more effort from the research team to ensure that parents were involved and the review was rigorous. The research centre (C7) had an established PPI system (C6) including a funded co-ordinator who supported parents and undertook extensive outreach work. The centre was also part of a CLAHRC with a vibrant PPI community and resources for PPI (C4), and parents were reimbursed for their time and received training (C2) for their role. Resources also included the time researchers needed to work with parents on the systematic review. This level of resources was necessary for an entwined model of PPI (see Chapter 7). There was a strong focus on positive relationships (C5) that were developed over time and enhanced by social activities. This led to a positive experience of PPI (C8) for all involved and the sustaining of a virtuous cycle whereby PPI became increasingly embedded. At the start of data collection PPI was already embedded in CS21, so there was no change in the tracking radar plots over time.
The need for time to develop a PPI infrastructure that could support embedded PPI was also clear in CS22 (Box 7). The topic area of CF had little history of established PPI structures, and the study designs are more commonly towards basic science. This case study captured the early days of study set-up and developing PPI, so radar plots were less full. After a year and despite focused work by a PPI champion, the radar plots remained unchanged, suggesting that embedding PPI requires a significant amount of time: at least 5 years. The limited impact of PPI may reflect the very early stages of a research study; however, as other case studies show significant impact in the early stages of the research cycle, it may also be indicative of a neophyte PPI structure which is not enabling a fuller impact.
Case study 22: CF Parents of children with CF (one member of charity). Experience of committees.
Outsourcing patient and public involvement
For case studies not hosted by organisations with existing PPI structures, an alternative was to recruit an ad hoc, project-specific group of people or individuals to support the study as PPI. However, another possibility was for research teams to access external PPI groups or panels. Topic area (C3) was an enabling factor and respondents from two national networks covering dementia and diabetes mellitus both held a list of names of interested PPI representatives. One network had a formal registration process on its website (which was updated annually and had around 3000 names) and the other spoke of a more informal, dynamic list based on personal relationships. Both networks had ready access to around 150–250 regularly involved and engaged PPI representatives; had been approached by researchers (in this topic area and beyond) for PPI in protocol development, funding before the application stage, help with PPI budgets and recruitment; and often organised local focus groups to gather PPI views.
Panels were preferred by some because they were more focused on a specific question and were hoped to consult with a wider population:
I think that the PPI, you get from a reference panel is potentially more focused on specific important questions . . . for example a Trial Steering Committee which might only meet twice a year or, and if the layperson can’t attend.
Panels were felt to be a very effective way of using PPI time and skills, and easy access to existing groups made PPI feasible within the time line of a project. However, although convenient, recruiting solely through local PPI networks with trained and prepared members of the public was seen as not always appropriate for effective PPI. Researchers within some of our case studies expressed concern about the difficulty of recruiting more than just the ‘usual suspects’:
one of the problems is ensuring that you actually are getting real and representative user involvement and not just the people who happen to be around who always get signed up to these user involvement things . . . actually it’s quite easy I think to get the usual suspects.
There was also evidence that relying on an external group had led to a ‘one-off’ model of PPI in which researchers had requested help for just one task and there was an absence of PPI throughout the rest of the research process. This limited the potential impact of PPI and was characterised by a more ambivalent view towards the value or impact of PPI. In a few case studies, the CI appeared unconvinced of the value of PPI:
but where’s the evidence that it [PPI] does work, maybe there’s scientific management for a programme that should just be done by scientists, internal, external and managers and maybe there are other elements that really need a PPI input, I don’t think that’s terribly well worked out at the moment.
Case study 01 (Box 8) provides an example of this model.
Case study 01: dementia Panel: an established local research network panel with a paid co-ordinator, who has worked with CI before. Panel asked for views on access to patient notes.
The evidence suggested that training around PPI for researchers was likely to be an enabling context, but there were mixed and even polarised views on training for lay representatives. Nevertheless, even in case studies with embedded PPI, researchers had not received formal training on PPI. Rather, they had learned through experience within an environment supportive of PPI.
Understanding PPI as different from study participation was a prerequisite for the development of appropriate structures, purposes and roles for PPI. Most researchers could articulate the difference; however, it was significant that many junior researchers could not. This lack of understanding was attributed by some to a lack of formal training:
there was no formal training, I just, you know, from what my colleagues and supervisors have just briefly mentioned but nothing formal on it and I think it would be an important part early on for trainees interested in research.
Of those lay respondents in the case studies who had received training, the majority had been provided by CLAHRCs, public involvement groups or research networks. However, the need for training for lay representatives did not emerge so strongly from the data as for early-career researchers. Indeed, various arguments were put forward against training, such as potentially compromising the lay perspective, or skills already existing because of extensive PPI experience or transferable skills from work life:
I don’t think there was any necessity for training at all. There’s certainly obviously one or two people have got good committee experience who are sitting round the table, on the lay side, so no, I don’t think there was any necessity at all.
In stark contrast a minority of lay representatives suggested it was impossible to contribute meaningfully without some understanding of research:
I don’t think that you can just so easily shove inexperienced patients in to a PPI role without them being, having some training and having some support because there’s no question that, you know, the whole research agenda is incredibly complicated and they talk in a language which is, you know, if you’re not a researcher you sometimes have no idea what they are talking about when they’re talking about face validity and constructs and confidence intervals and so on and so on.
However, the above quote suggests that this lay representative felt it was essential for the PPI representative to be able to cope with the research world, rather than the research community needing to make the effort to include the lay world in an accessible way. Making the effort also included working towards recruiting lay representatives who reflected the diversity of a study population.
Recruiting lay representatives reflecting the diversity of a study population
Table 13 illustrates the CMO configuration for this action. Formal training for researchers played a part in helping them to identify the need for this action, but experience on the job of recruiting diverse lay representatives was a more common enabling factor. It was easier to achieve this action when the research setting interfaced with and had access to a diverse population. Particular topic areas such as IDD had developed links with less-heard groups, and in a number of case studies these links were provided by a PPI co-ordinator or a lay person within an outreach model. In a few case studies, recruiting lay members as representative of the study population was actually operationalised through recruiting study participants to also have advisory input: a dual role.
Context, mechanism and outcome configuration: diversity
Strategies for recruiting lay representatives
Being able to understand the research through the viewpoint of a target population was seen as key:
I was sat on a table with speech and language therapist, physiotherapists, paediatricians, and a handful of parents, all as equals, you know? . . . and I was just thinking ‘What does that mean to me as a parent’, thinking of my child’s capabilities and all the other children that I knew.
However, finding lay representatives who they felt could present the viewpoint of the research study’s target population was often challenging for research teams. This was more frequently cited for BME groups. We attempted to include case studies that were likely to have a target population from the BME community. One case study specifically focused on the experiences of a particular ethnic group living with arthritis (CS18). One of the study sites was located in an area with a high percentage of people from this ethnic background, and the lay representative was recruited through that site:
I was going to the clinic, attending the clinic and the nurse actually asked me if it’s okay because I speak different, three or four different languages.
This opportunistic recruitment meant that CS18 benefited from the input of a lay representative who demonstrably understood the enablers of recruitment in far more depth than the research team:
what we had was an audio CD because a lot of people do not read the script, [specific language], so some of our interviewees did not speak English so we got the information sheet translated and put onto CD and she [PPI01] listened to that and gave feedback on that because there are different dialects of [specific language], apparently some are deemed posher than others and there’s all sorts of things that I was not aware of initially.
While there was trust in this lay representative’s expertise demonstrated by the peak in the collective action segment of the radar plot (bold outline) (Box 9), the construct as a whole was affected by one major challenge: that the CI (who was effectively the PPI lead within this study) and the PPI representative were located at a significant distance from each other.
Case study 18: arthritis Patient and public involvement representatives normally recruited to each study via an established PPI group based at the university. Only one took part in CS18. Expert by experience (has condition). This PPI representative was (more...)
We also included four case studies focused on type 2 diabetes mellitus with the expectation that these would include a relatively high percentage of research participants from BME groups susceptible to this condition. One of these case studies (CS03) (Box 10) had used strategies (advertising and interviewing applicants) to recruit a diverse range of lay representatives:
Strategies used when recruiting individual PPI representatives reflected researchers’ attempts to involve people who could bring both specific and broader perspectives. Recruitment of individual lay representatives was from five main groups: general public; expert by experience; carer/parent; patient representative; study participant. Individuals could take on multiple roles and represent their views at different levels. However, relying on just one or two individuals also raised the issue of representativeness:
it’s impossible, you can’t expect one person to speak on behalf of everyone so I guess the question would be what would be useful, or truly influential patient involvement? Is it enough to have just one person or two people who will be very, very good and who will be enthusiastic, but the question is, how representative are their opinions into their input?
Nevertheless, one model of using a single lay representative as a conduit to a broader constituency appeared to work well. The outreach model, as exemplified by CS02 (Box 11), involved one representative from a charity who then linked with 20 members of the support group in an iterative way to inform the study design and recruitment materials. An unforeseen impact from this PPI was to raise concerns over the safety of the study, resulting in extra study visits and a questionnaire (see Impact and outcomes of patient and public involvement).
Case study 02: diabetes mellitus Steering committee: national representative. One PPI representative (senior position in charity) sits on trial steering committee. PPI representative then consults the (20) panel members, invites feedback, collates answers (more...)
Some case studies also bought together project-specific panels. Case study 12 (Box 12) was particularly enabled by being located in a teaching hospital with relatively good resources for research (C4), which were sufficient for PPI honoraria.
Case study 12: diabetes mellitus Newly formed panel: experts by experience, participants on the trial, mentors on the trial. Less PPI at the early stages of the research, became involved with study documents. One lay representative had already worked (more...)
Challenges in ensuring diversity
Researchers acknowledged there was much work to be done to ensure diversity in PPI. The fact that only a very few of our PPI respondents were not from a white British background exemplifies the common problem as described by one of the funders:
we have as much of a problem as anyone about, you know, diversity and all those issues are still issues which we need to explore further. I think often public, people who get involved with PPI are a self-selecting group and it’s really difficult to bring in groups wider and that’s a usual excuse and I hate it, and I hate it when researchers say it. But I, it, you know, for example if somebody couldn’t read and write how would we include them in this process? If someone wasn’t, we do manage to include people who are not computer literate but then these days how are they going to even find out about us if they don’t have a computer? How are they going to know who to ring?
Difficulties in accessing ‘representative’ PPI were particularly marked for public health studies when the focus was on healthy or ‘at-risk’ populations with particular lifestyles. The difficulties for public health studies was also linked to the setting (e.g. broad communities) in which they took place. Those studies taking place in settings with ready access to patients obviously had greater potential for recruiting both participants and PPI representatives onto their studies:
the problem at the university is that those researchers that aren’t clinical or don’t have good links into the clinical setting, it becomes very difficult to identify service users to sit on a panel like that so we’re in quite a privileged position because we’re right next to the outpatients clinic and that makes our life a lot easier for maintaining that model.
The dual role
Clinicians who were also researchers had the added advantage of having ready access to patients as potential PPI representatives. For example, one case study (CS14) (see Box 26) hosted by a district general hospital with few embedded PPI structures was able to involve patients through an active condition-specific support group facilitated by clinical department staff. However, the distinction between being involved and being a research participant was often blurred in this case study, with PPI being understood by many of the respondents as a dual role. This dual role was overt in another arthritis-related study (CS08) (Box 13), where the PPI respondent was also a study participant. While there are potential ethical conflicts here, this lay representative actively recruited participants through her own network and had a strong sense of purpose. This action-orientated approach to PPI resulted in a fuller quadrant in the radar plot denoting a positive appraisal of the PPI within CS08 (see bold outline in box figure).
Case study 14: arthritis Hybrid: national representative, local service user group and expert by experience. However, these components did not come together and hence there was no cross-fertilisation. The national representative has advised nationally (more...)
Case study 08: arthritis Dual role: PPI representative already known to CI through being on funding committee but also has joint issues so is participating in the research as well as acting as an advisor.
Although respondents in CS08 were comfortable with this dual role, it was operationalised in an ad hoc manner that was unlikely to lead to any embedding of PPI. In contrast, this dual role was carefully constructed and managed in a longitudinal cohort study (CS13, see Box 16). A rolling PPI panel was drawn from study participants and the role was clearly defined and separated. The impact of this role included both increased public engagement with the study and improved participant retention. This model of PPI was enabled by topic area (public health with healthy participants) and the longitudinal design, which allowed relationships to be developed and sustained over time. This was a well-resourced study with dedicated PPI staff and good organisation, and was run from community hospitals at the heart of local populations. For the public undertaking this dual role, there was a sense of ownership of the study, with subsequent sustained engagement. Two tissue bank studies also had lay representatives who advised but also had donated samples. In CS17 (see Box 23), engagement was sustained, but for the other (CS16, see Box 20) the sense of ownership became a cause of conflict, which is presented in Enablers and barriers to trust.
Case study 13: public health ‘Consultation’ panel hybrid model, built upon earlier, less formalised (mass consultation, participant feedback) PPI panel drawn from large cohort study (now mostly retirement age). Panel meet quarterly with (more...)
Case study 17: diabetes mellitus Established panel for over 10 years; about 30 on panel and two attend each monthly meeting (usually invite four). Meeting and reviewing papers. PPI mixture of research-naive and ‘professional lay people’. (more...)
Case study 16: diabetes mellitus National expert network: charity. Dual role (participants and representatives).
If this dual role was to have a positive impact, then the purpose, structure and clear role boundaries needed to be defined.
Whole research team is engaged with patient and public involvement
A distinct action of case studies where PPI was embedded was engagement of the whole team with PPI, and having the skill set to do this. Funder requirements (C1) for plain English summaries were bringing into sharp focus the need to be able to explain technical aspects of the research in an accessible way, and the most effective way of doing this was to work in partnership with lay representatives (C5). This needed time to achieve (C4) and could also be helped through specific training (C2). It was particularly evident as an action in the IDD topic area (C3). The CMO configuration for this action is shown in Table 14.
Context, mechanism and outcome configuration: whole research team engagement
Relying on one person to lead patient and public involvement
As described in Purpose, structure and role of patient and public involvement, having someone to lead and co-ordinate was important in embedding PPI. If there was not a designated co-ordinator, this role was often carried out in addition to a junior researcher role and was not part of the original job description. Different levels of commitment to this role were found and PPI representatives spoke of often not having one ‘designated person’. Continuity of personnel with responsibility for PPI was an issue. For example, one PPI representative reported seeing someone different at each meeting (CS05) (Box 14) and another said the enthusiastic support ‘gradually dwindled’ (CS14). If the whole team was not engaged with PPI, the loss of the PPI champion had a detrimental impact.
Case study 05: arthritis Steering committee: one expert by experience, recruited through national arthritis body, who was a patient of the CI. Was asked to change consultants. Nursing background and interested in research. Understands some of the medical (more...)
Patient and public involvement as a collective action
Case studies demonstrating agreement of all individuals in the team that PPI should be part of their work were particularly strong within the IDD topic area, where there is an acknowledged history and strong investment in PPI. The CI from CS11 (Box 15) clearly articulated this:
Case study 11: IDD Two PPI representatives are new to PPI in research (one voluntary worker, one carer) and one more experienced (expert patient). Preference for PPI recruitment from established condition-specific networks (built on previous relationships (more...)
in order to understand and develop theory effectively, you need to really have a conversation with service users and probably somebody who has the problem with the condition, and that is how psychological theory develops. You know, it is that, ‘Are you doing something to people or are you doing it with people?’ and doing it to people, you know, there’s such a power imbalance, and given the backdrop and the history associated with people with disabilities internationally, that’s not politically palatable to me personally and to, you know, society, I suppose.
Although the NIHR funding (C1) was used to pay out-of-pocket expenses, this case study was hosted by a medical school, with the CI reporting limited engagement with PPI. However, it is the topic area (C3) that is the strongest enabling feature, suggesting a culture where ‘nothing about me without me’ shaped research practice. This CI also framed the challenge to explain technicalities more clearly to lay audiences positively, as good practice for communicating ideas to the public:
saying things like, ‘Oh, this is a stepped wedge design,’ and ‘We’re randomising,’ . . . you can use that language, but people aren’t going to understand it. So, you’re going to spend all your time explaining what you mean, so you might as well just use simpler language to start with, and perhaps really as a researcher or scientist, that’s a good thing because it helps improve your communication skills with lay people and the general public.
This whole team engagement was not unique to IDD as a topic area. For example, in CS13 (public health) (Box 16) although a project-specific PPI panel was facilitated by a lead person, all senior researchers took it in turns to attend panel meetings. The embedding of PPI in this study was enabled through access to resources from grant monies (C4) and a longitudinal design (C3) that enabled relationships to be sustained (C5).
Mutual understanding and trust
While formal training (C2) had a part to play in helping both researchers and the public to understand each other’s roles in research, positive relationships (C5) sustained over time led to PPI as a positive experience (C8) and the development of trust, which was key for this action. Reimbursement of out-of-pocket expenses and time contributed to maintaining trust. If clearly articulated, funders’ expectations (C1) of reimbursement were an enabling context, as was the topic area (C3) if reimbursement was normal practice. Host organisations (C7) had a key role in providing resources (C4) for reimbursement and ensuring payment was smooth. The CMO configuration for this action is shown in Table 15.
Context, mechanism and outcome configuration: mutual understanding and trust
Coherence in understanding the contribution of patient and public involvement
As the bedrock for trust, lay representatives and researchers needed to have a shared understanding of the purpose of PPI. Novice researchers were more likely to be unable to fully distinguish PPI as separate from research participation. For example, a registrar in CS14 defined PPI as:
patients contributing to research and enrolling into research studies, whether they had portfolio status or not.
While this impeded an understanding of what PPI could offer to the research process, other researchers and PPI representatives provided a variety of reasons for carrying out PPI including gaining insight into the lived experience of a particular health condition, and to provide reassurance for researchers that their research question resonates with the patient community. This was exemplified in CS07 (Box 17).
Case study 07: diabetes mellitus Experts by experience panel (≥ 10 patients who have lived with diabetes mellitus for over 5 years). Recruited from diabetes research network panel. Members of the CS07 panel are on their own Diabetes UK (more...)
Case study 07 was enabled through an established diabetes (C3) network group (C6) housed in the host organisation (C7). The study focus on processes of care (C3) also enabled coherence. PPI members met regularly and were involved in a number of projects which helped promote sustained engagement (C5). However, lay representatives were not reimbursed for their time because of bureaucratic difficulties; rather, they were taken out for a social meal.
Whereas the experience of established panels was seen by many researchers as a bonus, others spoke of the tension between wanting someone who was ‘outside’ the research environment and wanting someone who was also able to engage with research studies (possessing some research skills) and thus perceived as being able to perform the necessary PPI tasks. This notion of wanting the right person with a short distance between direct experience and its interpretation but who also can feel comfortable in a research environment is maybe a contradiction in itself. There was evidence of an ongoing debate about whether or not the experience of a condition (direct or indirect) was seen as less legitimate because of that person’s professional background, experience or training in research or health care:
we’re all patients . . . yeah, and some of us have PhDs and some of us don’t, so it’s quite difficult to kind of define what is a lay representative . . . they’re really just people who, actually, through their qualifications, they should be research assistants but don’t happen to have a job in research but are on a lay panel. And I kind of think ‘what . . . are you really a lay person? You’re just an unpaid researcher’, but I’m not entirely sure whether that’s right . . .
Our findings suggested that defining PPI as a discrete and commonly understood component of research was difficult but the range of PPI representatives can be summarised through a fourfold typology based on an insider/outsider perspective (on one axis) and a range of research skills (on the other) (Figure 8). Research studies can be placed along these axes; however, within one research study there may be a mixture of individuals with varying skills and perspectives.
While the insider/outsider debate was articulated in a number of case studies, those that had embedded PPI were likely to have sustained engagement with the same group of PPI representatives over a long time.
Building relationships over time
We found that the relationship between the researchers and PPI representatives was a key factor for successful PPI. Relationships took time to establish, which was particularly difficult at the beginning of research (or even before the proposal is written). One finding from the case studies was that the majority of PPI representatives had a previous working relationship with the research team. Of the 51 PPI representatives, 33 (64.7%) had some prior working relationship with either the CI or members of the research team (three were patients of the CI and also members of a service user group, so had worked together before), nine (17.6%) had a more distant acquaintance (but described themselves as being within the same loose network) and nine (17.6%) PPI representatives were completely new to the research team (recruited from outside existing structures). The obvious advantage of having a previous relationship meant that PPI could start as soon as possible within the research project, whereas more time was needed to establish relationships through informal small meetings if the lay representatives and researchers had not previously met.
Research teams spoke of initial meetings where everyone got to know each other and having these times of informality before starting on the ‘academic’ business. The benefits of PPI representatives having the opportunity to get to know each other also provided support later in the project. Social elements were important in encouraging and maintaining continual engagement in the research study and should not be underestimated by researchers. Some of the case studies also ran social events outside the research, which again helped to build and maintain relationships:
We do these family fun days as well, which we get together once a year, just to get together at the zoo or something. [PPI co-ordinator] works phenomenally hard to build relationships with the families that . . . So, it’s a partnership really.
In more formal advisory or steering committee meetings, PPI representatives spoke of being specifically welcomed into the meeting (sometimes being met beforehand). Most importantly, during the meeting, successful PPI was being ‘genuinely’ listened to and their comments taken on board. The relationship between the researchers and the PPI representatives was key to fully embedding PPI within the research study. The researchers needed certain social skills, which included being approachable, good listening skills and group work skills:
initially facilitating and always presented a positive, welcoming, friendly, supportive space to you.
Patient and public involvement representatives reported that meetings needed to have a ‘flat structure’ where PPI representatives felt free and at ease to speak.
Relationships could change over time, either through loss of trust or as trust developed, as illustrated in CS19 (Box 18). Although only subtly different, the radar plots show a change in all quadrants but particularly in collective action (bold box), where M10 (trust in each other’s PPI work and expertise is maintained) is included as a mechanism. This reflects clarification of roles and the improving relationship and trust in the PPI work:
Case study 19: public health Committees: two representatives (one a coapplicant) on trial steering committee (meet twice a year). Two PPI representatives on trial management group (meet once/twice a year).
I don’t know what they want me to prepare for, I don’t know what they want me to do, and I don’t know if the confusion is because of my professional role [. . .] and I don’t know if they’re waiting for me to take the lead, and I’m waiting for them to treat me as a patient. . . . I’m a little bit disappointed. It feels almost tokenistic.
Conflict between roles resolved . . . CS19PPI02 feels happier now that they are no longer post-surgery, less vulnerable. They had a long chat with a rheumatology person in department who had done a lot of PPI work who reassured CS19PPI02 that one PPI voice is better than none, more confidence to feel you don’t have to represent all patients, can just give your own experience. Different way of looking at the value of their contribution.
CS19PPI02, notes of tracking interview 6 months later
Trust as the foundation for patient and public involvement
A relationship framed by trust was the minimum requirement for PPI to have a positive impact. Although PPI was not embedded within CS14 (see Box 26), the relationship between lay representatives and the clinician was based on trust built up from a clinician–patient relationship with continuity over time:
there’s a trust there, I trust them that they wouldn’t ask me to do something that wouldn’t be, you know, it would be difficult or wouldn’t necessarily be appropriate or whatever.
Some researchers also trusted their lay representatives to undertake the work in a way that was workable for the researcher:
It’s always good, you know, on research it’s you need good people you can work with and that’s what I would be looking for, to set up some relationship of trust and mutual respect . . . Without thinking of them as a hindrance.
Case study 15 (Box 19) exemplified how, even when resources were minimal for PPI, the very existence of mutual trust resulted in positive PPI impact. This study was housed in an organisation with no PPI infrastructure and with minimal resources for PPI despite the study being funded by the NIHR. The research setting [intensive care unit (ICU)] provided particular challenges: consent and recruitment difficulties in a high-stress environment; difficulty in recruiting lay representatives, as they were often traumatised by their time in ICU; and lack of resources, as there was no large charitable funder. The lay representative in CS15 had an impact on recruitment processes for the study and provided a valid argument to present to the ethics committee for this approach:
Case study 15: dementia One national representative. PPI role confined to trial steering group.
we put a thing here saying in the consent ‘to continue information now you’ve regained consciousness, we would like your permission to continue in the research’, now [PPI01 has] written ‘I realise this might not be possible but in my own experience of delirium I was regarded as having capacity long before I was in my right mind or capable of rational decision making. I appreciate this may be beyond the scope of the study but if there were any way to change the emphasis for the patient remaining in the study unless they chose to opt out, I think that would be much better’.
It can be clearly seen how M10 appears as a peak in the collective action section of the radar plot (bold square).
Enablers and barriers to trust
Sustained feedback to PPI representatives was a clear enabler of trust and continued engagement:
and they’re always thanking us for our participation. All the time. Always saying, you know, I get it in e-mail messages, we get it at the meetings and they always say how valuable our input is, you know, they make us feel worthwhile and that is good.
This feedback was recognised by many researchers as being important:
I think it’s important, what I’ve learnt is that if you are going to ask them things you need to feed back to them so that it’s terrible to actually ask their opinion and they never hear again, so I’m quite good now, it’s an agenda item that you know, I feed back what I’ve heard from different people, because they’re left wondering . . .
The forum in which researchers and lay representatives came together was also an important enabling context for trust. The majority of our case studies had PPI representatives as members of steering committees (one or more lay representatives per study) which met every 3 or 6 months to advise the research team but did not necessarily have a ‘hands-on’ role with the research. However, different research teams made different uses of their steering committee members, with some members making comments only during the meeting and others who were asked to comment on documents throughout the study. As shown in CS04 (see Box 22), there was a varying sense of equality within the committee:
Case study 04: diabetes mellitus Experts by experience.
it wasn’t run by the Professor from the top down in sort of military fashion, it was a round table discussion where anybody could contribute . . . whilst it could have been intimidating actually it was very welcoming, and the person who chaired the meeting was very sensitive to that.
However, the formal term ‘steering committee’ put some PPI representatives off: ‘if they said someone to be on the Steering Group I mean I would never, I would have just thought well that wouldn’t be . . . that sounds too high powered for me’ (CS04PPI01-01).
Despite it often being seen as good practice, there was little evidence that this relational integration was being formalised through job descriptions or contracts for the PPI representatives. However, at the start of the study researchers often clarified expected commitments to attending meetings; other PPI input was often less formalised. Those case studies which had high levels of PPI activity could achieve this through flexibility and allowing PPI representatives to ‘dip in and out’ as their other commitments also varied:
parents will stick with the programme because they know that they can step in and out if needs be, and the team also think about how they can include people that might not be able to travel, so whether it’s joining in a meeting by phoning in to it and participating over the phone or sending responses or comments and feedback in an e-mail, so it’s made it available and it’s made accessible.
However, trust in CS16 (Box 20) was lost because of a lack of formal agreements about access and ownership of a database. This issue appeared midway through data collection and the impact can be quite clearly seen on the radar plots tracked over time. The impact can also be seen in the way the PPI was appraised (hatched box); because of this experience the charity would get legal documents drawn up before working with researchers again.
Virtual patient and public involvement
There has been a recent move towards the increasing use of technology and teleconferences being used as a means to include lay representatives who might otherwise not be able to attend meetings. However, this move towards ‘virtual’ contact as opposed to ‘face to face’ has implications for the establishment of relationships. One lay representative said that they always wanted an initial face-to-face meeting to make any following (virtual) meetings easier. As presented in Strategies for recruiting lay representatives, being unable to meet in person also caused problems in CS18, where the CI and lay representative were in different regions. The challenges of building and sustaining relationships through virtual contact were also raised by a funder. Maintaining arm’s length relationships was acknowledged to be difficult for the army of remote lay reviewers, and they found it difficult to sustain any more active involvement. Case study 23 (Box 21) provided an example of virtual PPI with young adults, and also demonstrated how this medium provided an outreach model (see Discussion).
The benefit of this virtual form of PPI was obvious:
I’ve got a main informant on Twitter . . . asked her if she could work with me on developing a paper and she’s a young person . . . and so she basically threw some question out to Twitter to her one and a half thousand followers . . . and we sort of put some questions to them and we’d written a paper within 3 weeks.
Case study 23 also had an advisory PPI group made up of four young people with the condition. At their request, this group was hosted separately from the research team meetings and deliberately had an ‘informal feel’. These separate meetings were envisaged to be a series of evening, face-to-face meetings with monies allocated to cover travel expenses. However, in reality, as a result of competing demands placed on the attention of this age group, the group did PPI ‘virtually’. Co-ordinating such involvement was a significant workload, which was difficult to predict and cost for. However, as this was adapted to over time, the tracking radar plot became fuller.
Reimbursing lay representatives for their time is intuitively an enabler of trust and seen as normal good practice, yet payment for time was surprisingly rare in the case studies, although all were paid for out-of-pocket expenses. This was not only because of financial restraints but also reflected a continuing debate from a minority of respondents on how payment fitted with the notion of volunteerism. For example, in CS08 (see Box 13) PPI was felt to be more of a voluntary role, which payment would then ‘professionalise’:
No, I think it should be voluntary . . . because you’ll be paying for services for like, I guess, advice or public feedback, if you’re paying them then it’s, I don’t think, it’s more official, I guess . . .
This was rejected by many lay representatives:
It is and part of the problem is that people just kind of make an assumption, oh they’re patients, we won’t need to pay them. And you know our time in our life is just as . . . important as a researcher’s or a doctor’s.
Among the five case studies reimbursing for time, the amounts ranged from £25 per meeting to £75. This was often enabled by the host organisation, such as a CLAHRC or research centre. However, even within well-funded host organisations such as HEIs, researchers related institutional barriers to paying lay representatives within institutional systems reliant upon inappropriate standardised status classifications (such as lay people having to be classed as visiting lecturers), electronic forms and difficulties dealing with cash refunds, as well as payments affecting lay volunteers’ benefit payments and tax arrangements. Hence, the host organisations that managed reimbursement and arrangements seamlessly made lay representatives feel a collegial partner with the organisation.
National charity representatives reported that they were being asked to be involved with an increasing number of research studies, which is not necessarily the main part of their work. One PPI representative called for there to be some recognition of cost to the organisation and the consequences:
there must be other charities like me in other therapeutic areas who are getting the same issues . . . So I would have thought that we should be looking at this at a national level to say if patients are going to be involved in research in a way that they have not been previously then they need support to do that and it should not, all of the cost of doing that, should not fall solely on the third sector. We should be getting some kind of NHS/government DH [Department of Health] support for this.
One paid charity representative said her workload was such that she did PPI work in her own time. Perhaps in acknowledgement of this, another charity representative in a PPI role was contracted (part time) by the research project – yet further blurring the lines between PPI and researcher. It could also be argued that paid employment also introduces different obligations and commitments to involvement, although it has the potential to clarify an otherwise open-ended commitment.
Opportunities for patient and public involvement throughout the research process
A characteristic of studies with fully embedded PPI was that opportunities for PPI in all parts of the research process were fully exploited. The CMO configuration for this action is shown in Table 16. As an enabling context for embeddedness, it was not necessarily how the PPI was organised but how much PPI representatives were encouraged to be involved and were exposed to different areas of the study throughout its stages where they could have an impact. There were reservations from some researchers on the potential for PPI in the early work of basic science designs (C3). However, we found that, with appropriate training (C2) and experience, lay representatives were effectively contributing to funding decisions for basic science studies, and prioritisation for tissue bank studies. This was further enabled by having a pool of lay representatives (C6) with sufficient interest and expertise to contribute effectively at all stages of the research.
Context, mechanism and outcome configuration: opportunities for patient and public involvement throughout the research process
Patient and public involvement and the research cycle
Performing the necessary tasks for PPI appeared limited to certain stages of the research cycle. Some researchers recognised the importance of having PPI before studies were funded but acknowledged the difficulty of finding appropriate PPI representatives in a short time frame. Many researchers felt comfortable involving PPI representatives in the general review of study documents but not so certain about involvement in other parts of the research process. In most case studies there was significant PPI during study set-up, but this often decreased as the research progressed, for example when patient recruitment or data collection started. Researchers often reported that the pressure to recruit research participants (during data collection) resulted in less capacity to enable involvement of PPI representatives. This impacted most on relational aspects, with changes evident in this sector of the NPT radar plots.
The distinctive characteristic of case studies that enabled involvement at all stages of the research process was the presence of a dedicated individual facilitating PPI (e.g. CS21, see Box 6). A complementary or alternative approach was having a lay coapplicant who was fully involved from the inception of the design, and sometimes undertook an outreach role, or worked as an integral part of the research team. Case study 04 (Box 22) had one experienced lay member as coapplicant; this was enabled by the research topic being focused on a patient evaluation tool, and by having an established PPI group with experienced lay members.
Patient and public involvement in different research designs
Funders and some researchers espoused stereotypical assumptions about the limitations of PPI in certain research designs, such as basic science, which has traditionally been an area with little PPI. A PPI co-ordinator working in a medical setting and talking about CTIMP (clinical trial of an investigational medicinal product) research observed that, because some researchers believed patients have difficulty in understanding the science and bench side of research, they find it hard to engage and have meaningful PPI. Similarly, one funder suggested PPI was more compatible with applied science:
I think it comes down to the complexity of science and the type of research that you’re talking about, some studies: when it’s very kind of lab based and it’s very molecular, doesn’t really lend itself to being able to involve the public or patient, you know, at a very kind of technical level. But there are a lot of opportunities in medical research where it is viable and it is possible to do it.
Other researchers also felt that involving PPI representatives in ‘technical’ discussions about certain research designs was tokenistic, as they were able to contribute very little:
I mean we’re doing a review of reviews . . . so it’s methodologically very challenging, it’s conceptually quite challenging, half the academics feel out of their depth, intellectually and conceptually. The PPI feel . . . that’s not to denigrate them or to put them down in any way at all, it’s not what PPI is about, . . . No, they come to the steering group and then they feel a bit left out and I think it’s a shame, I think it’s, you know I think it’s disrespectful in a way that not having, I mean I think, for that project it would be more respectful not to have PPI, does that make sense?
There was also some evidence that researchers felt concerned that some PPI representatives who lacked scientific understanding could jeopardise a study at the design stage. However, another funder challenged assumptions about the ability of experienced PPI to review less applied, more basic science applications:
when we set up these committees, there was a fear or a worry that they would have trouble reviewing basic science applications and therefore they would be more drawn towards the clinical ones, but actually what happens is, it’s quite funny really, it’s almost the other way round. I think they can see the pipedream or the hope at the end, and almost they’re more critical about clinical studies, I think maybe because it’s closer to the patient benefit.
Indeed CS17 (Box 23) successfully involved PPI in approving bench research proposals in genetic epidemiology through a panel model whereby the PPI representatives are presented with lay protocols written specifically for the panel.