Improving Research Inclusion: learning from NIHR and Research Council funded studies in England

Patient and Public Involvement

In this paper we summarised the learning from these four projects, rather than report fully the PPI arrangements of each project. In brief, in ATTUNE young people and lived experience expertise participated in the funding application and design, and in the delivery of each component, including deliberations on ethics, and in the interpretation and writing up of results. In Co-Pics and the related PhD project (both still being written up) on multiple long term conditions including psychosis, people with lived experience reviewed and contributed to and were involved in the application, in the data collection, interpretation, and writing up. In the NIHR BRC themes on Preventing Multimorbidity (in which Co-Pics is adopted) and the Developmental Theme, we have worked with lived experience experts with the relevant experiences and conditions to inform how we should undertake research; in these studies we had to devise the engagement and involvement plans after making contact with potential participants, showing flexibility and local and group specific adjustments in our approach. Again, people with lived experience were and will be involved in interpretation, for the NIHR BRC Developmental Theme. For this paper, we did not establish a separate PPI group, although we did share a summary of the paper and our main proposals (the checklist) with a panel of 5 young people, asking them to rank our proposals in priority. The studies were mostly following an experience based co design methodology, so mostly were not trials. The co-designed interventions are being or will be evaluated in the future. Where we outcome measures in ATTUNE, which were recommended by the funder to promote comparison across studies, but were also reviewed and critiqued by young people’s panels, to inform selection and modification and presentation.

Agreeing concepts, vocabularies and evidence

The conduct of research is often shrouded in technical terminology, either reflecting specific methodological paradigms or disciplinary framings of the problem that the researchers seek to solve. Esoteric or technical language is sometimes necessary, linked to theory to help us advance science, but such language can act as a barrier to inclusion⁹. For example, on interrogating concepts like personality disorder, or disease or illness, or ethnic group, agreeing an accessible vocabulary and explanations of causation often reveals conceptual confusion or obscured ambiguity amongst scholars. Such questioning helps improve the precision of the language used and exposes traditions and conveniences that must be revised. These negotiations ensure partnerships are grounded in shared concepts from inception, and that there is agreement on how words that carry power are to be used. Linked to language, once research is completed, the findings should also be accessible to the public, in a range of appropriate visual, audio, and sensory formats. For example, arts installations and exhibitions are an excellent vehicle for sharing research findings and for influencing practice and policy.

RI seeks to democratise knowledge and shows what research is being done for public accountability; RI also challenges the language, framing, specific methods, objectives, and design; thus greater involvement of those who would not usually participate in research can bring benefits to them¹⁰. In our work with marginalised groups and with professionals delivering health and social care, it is always surprising how little of the research evidence is known. Therefore, assumptions of what is known or not before co-design and participatory methods should be challenged. Providing evidence summaries early in the process, again in multiple modalities, can help balance power to optimise trust and communication.

Trust, power and participatory co-design

The design, delivery, knowledge mobilisation strategies, and impact pathways should be informed and shaped by the beneficiaries so that research is relevant, ethical, and more likely to be impactful. Only through dialogue, with participants and intended beneficiaries through specified collaborative processes, will knowledge production address epistemic injustices and public and participants will come to trust the research process and later findings¹¹. This should be in partnership with those expected to implement research findings in practice and policy.

Participatory RI can identify research questions of importance to the public and patients, alongside refining or challenging those proposed by funders and professionals. However, an equally important function is to critique or to deprioritise questions or procedures that might reinforce stigma and prejudice, or indeed, halt research interests that are too far removed from patient experience. Such critique can prevent wasting resources on unhelpful approaches. This shift to find solutions during the research requires time to explore and progress, and may be viewed as demanding and costly, yet additional resources (time and funds) should be anticipated in the research design². Implementing RI can be felt as disruptive of conventional research pipelines, but necessary to shift power away from research teams towards the beneficiaries. We found a re-think was often required in response to poor recruitment progress; pauses and perceived disruptions offered valuable opportunities to review research designs and underlying philosophical and conceptual confusions and assumptions. Lived experience perspectives often help uncover these assumptions.

In our approach to inclusive research practice, we made use of participatory methods, emphasising co-design and collaboration with the intended beneficiaries from inception¹². However, participatory methods are seldom used or reported¹³. Co-designed and collaborative research contrasts with traditional approaches, where funding priorities and study designs are often determined by governments, think tanks, charities, and learned societies and researchers. These usually also lock in a particular design, rather than respond to the ongoing and newly discovered needs of potential participants to ensure inclusion. Another benefit of co-design and participatory methods is that those expected to implement findings and wider public stakeholders become more informed of what matters to the intended beneficiaries, and what really is at stake for them. This prepares them to champion the implementation of research and anticipate which local regulatory and governance procedures need activation.

Ethical issues

Research inclusion often raises ethical questions and dilemmas when designing and delivering research. Adopting an inclusive research approach means upholding an ethical responsibility to ensure justice and fairness in research, where all groups have an opportunity to benefit from advancements in science, rather than a privileged subset. Globally, ethical dumping (the practice of researchers from high-income countries conducting unethical or exploitative practices in lower income countries) and helicopter research (not considering the impact on the community once the research is concluded) are being challenged and must be rigorously avoided and managed¹⁴.

One way to combat ethical violations is to ensure research processes to recruit participants and deliver research are developed with and subject to critique by intended beneficiaries. Oversight committees will need to be attentive to potential dilemmas. Including ethical dimensions in all programmes helps to intentionally deliberate upon unanticipated dilemmas and see these as opportunities to learn. For example, in ATTUNE, we designed participatory methods involving multiple modalities of creative arts and communications (verbal, audio, video, sensory) and different approaches to co-design; this was time consuming and required thoughtful and sensitive engagement with the intended beneficiaries throughout. These approaches may be challenged if they are less familiar to funders, researchers and ethics committees¹⁵. Therefore, an important task is to explain and upskill such committees; this is additional work researchers must anticipate and undertake. In addition, the costs of more participatory and paced progress must be met.

There are many examples of how research needs to be adapted to respond to ethical questions. For example, adequate support is required for people to enter research, especially if they are frail, vulnerable, or have experienced lifetime adversity and unfavourable socio-economic conditions; this requires more time to assess what kind of support is needed and to build trust; participants will need more support than is usual during and after the research. It is relatively easy, in our experience, for such groups to be systematically excluded by tight inclusion criteria or when the research infrastructure and teams do not have capacity to provide the necessary support. Judgements about suitability may carry inherent prejudices about people’s desire and ability to participate, often compounded by poor communication of the research aims and what participation involves. People who are experiencing poverty, mobility issues, as well as multiple health conditions require more time in travelling and entering and leaving research, and resources (for example, taxis may be required rather than public transport). There may be child-care needs to permit participation, and these too need resources and time. People with carer responsibilities will need support during the research process, especially if they are managing multiple social challenges like paying fuel bills, living in rural or distant areas, and if they need to prioritise immediate health and welfare appointments over research (theirs and the cared for person’s).

Young people in care or living in precarity may struggle with lengthy consent procedures¹⁶; they may also not wish to discuss research participation with parents or carers. Whilst researchers and regulators insist on assurances of uniform informed consent processes and of confidentiality, should young people living in precarity with chaotic and unconventional circumstances be excluded, risking neglect of their significant health, welfare, and social care needs? If they have troubled and chaotic lives, they may not be able to participate in tightly scheduled research, or in a consistent manner; research should not assume levels of literacy or ability to express experiences verbally; for example, many struggle to complete questionnaires that require significant periods of concentration, and ones which ask about deficits and traumas such as maltreatment that may be distressing and difficult to recall. Again, the ethical dilemma is that such young people may be excluded, so procedures and practices and ethical conventions may need to be revised. Who will take responsibility in this instance? Social care and local authorities are often responsible for safeguarding protocols and protections. Their participation in research is a priority in the UK currently, yet this requires a larger infrastructure for social care research.

The use of incentives in research is often questioned, yet for participants from poorer sections of society with multiple responsibilities and few assets, remuneration should reflect the time burden, and the emotional and cognitive demands involved in recruitment and participation, and practical issues (travel costs and time). Providing cash incentives may help, but can also compromise welfare and benefits, if the rules of welfare receipt specify limits to additional sources of income. Remuneration should also take account of time away from work, carer roles, and childcare costs.

There are other examples of ethical challenges: young people may choose social media communications, or digital methods, which are often not deemed to be adequately secure and not favourably viewed by ethics committees or sponsors. Yet, if this is their preferred form of communication, it may improve representation, in part through building trust and a more convenient form of easy communication. One of our studies used social media recruitment achieving 300 participants in a few months (NIHR Oxford Health BRC; whereas for other studies (ATTUNE) this was deemed too risky and not supported. Consequentially, greater flexibility is required in ethics and sponsor procedures; greater (time) investment is needed for research teams to understand participants’ lives and how to adapt the pre-specified research infrastructure as a study proceeds. Testing out different methods might also be included in future research designs.

The structural barriers that prevent use of health and social services, can be replicated in research. Innovative models can relocate research processes into community venues, ones that are more familiar to and trusted by participants; however, this means improving the infrastructure for research in the community in recognised safe spaces such as schools, charities, museums and art galleries¹⁷. Governance and assurance policies can become constraining and prohibitive, reinforcing structures of disadvantage. Therefore, we advocate for all research organisations and employers to review their policies and practices, on an ongoing basis, and perhaps even by each study, whilst evolving new procedures and practices. Watchful attention to potential for improvement should be welcomed, rather than seen to challenge or violate protocols and standard operating procedures. Advisory Boards and Data Monitoring and Ethics Committees are part of the current expected governance infrastructure in the UK; we found that raising the challenges and issues outlined at these committees can lead to productive opportunities to re-imagine inclusive research practice as studies proceed. Practically, revising procedures will require ethical re-review which can delay studies if the governance processes are themselves not sufficiently well resourced to reflect the demands of inclusive research.

Integrating research inclusion and PPI

Patient and Public Involvement (PPI) is a widely accepted practice in health research, usually with appropriate representation on advisory boards, in research teams, and amongst participants. Resourcing PPI well, commensurate with objectives, can protect against tokenistic inclusion, and extractive and exploitative forms of PPI. Building trust takes time and researchers need to guard against potential for exploiting knowledge rich communities. Despite the advancements in PPI frameworks, there remain areas of improvement especially regarding impact on the emerging evidence base. Many PPI panels consist of members with similar health conditions and from similar social contexts without much consideration of intersecting vulnerabilities (e.g., race/ethnicity, sex, gender, neurodiversity, sexual orientation, socioeconomic status, disability) that shape health outcomes^16,18–20. PPI needs to be adapted to facilitate inclusion of those located at the intersections of multiple disadvantages related to identities and socio-economic position. However, this usually ends up being undertaken on little resource and may be seen as unethical and unfair and ultimately fails to recruit intended participants. Furthermore, funder agencies may resist the additional expense, even if the proposals are fairer and appropriately challenge historically ineffective or exclusionary processes.

We found that researchers should avoid rigid orthodoxies in PPI and EDI. They should build in additional thinking and time to consider the most marginalised in their localities and reflect on their own awareness and agility in grasping and overcoming knowledge gaps as a core task. Sometimes the pressure of preparing and launching a new research project against a strict timeline and working through all the contractual, ethical, workforce, and administrative stages can generate strain in the research team even if they have good intentions to make an impact. This needs care of the research team, especially if lived experience is prominent as this can be emotionally and cognitively demanding and place additional stress on teams. Building in reflective debriefs, and careful use of holidays and health days, alongside flexible working, and supports where required can be helpful. Indeed, research teams also often carry many undisclosed experiences of poor mental and physical health, and so the content of the research may be found to be distressing and emotionally challenging. A good research culture should be able to accommodate discussion of such issues, for example, by agreeing from inception a values framework, and reflective discussion. External facilitation can help team dynamics also. We recommend all members of the research team, irrespective of discipline, undertake research inclusion training, using and testing the participatory methods; for example, we’ve made good use of the approach recommended in living labs and learning labs. These ensure embodied and experiential learning rather than intellectual knowledge.

To improve RI, we compiled a checklist as a set of standards derived from our work on various research projects. Our desire is these standards are adopted, but different types of research may require tailored refinement (e.g. cultural adaptation of psychological interventions or developing randomised controlled trials).

Lived experience perspectives

Involving patients and lived experience experts is now common, but there are so many approaches and methods, that we propose these should always be co-designed specifically for each research project by those participating in that project and potential beneficiaries. The strategies we adopted and put forward evolved in partnership with project specific PPI panels, peer researchers, and community partners. We shared a summary of this paper and checklist with five young people through a survey. We did this to reduce burden to them of soliciting their views, recognising the paper is written as guidance for researchers rather than lived experience experts. We asked this group to rank our recommendations. We only present percentages ranking each statement as 1^st or 2^nd; see Checklist).