Review aim, questions and objectives
The aim of this review is to answer the over-arching question: What are the experiences of under-served populations with the informed consent process and its impact on decision-making about whether or not to participate in health research?
To develop and refine a programme theory identifying the contexts and mechanisms that affect communication of information and experience during the informed consent process, for under-served populations.
A further sub-set of questions identify the more specific aims to answer this question.
- What contextual factors influence the decision to take part in health research in under- served populations?
- How do under-served participants experience the information provided during the informed consent process in health research?
This review will involve stakeholders (described below) who will be invited to comment on the process of informed consent in health research as NIHR (Public) Research Champions, health professionals and clinical academics. NIHR (Public) Research Champions are volunteers whom are interested in raising awareness about health research, particularly to those whom often do not participate . This mix of stakeholders are considered to hold positions that are relevant to the topic of the review and to have a vested interest on behalf of under-served populations . Ethical approval is not required as stakeholders will be contributing collaboratively to content and dissemination, not providing data [36-38]. These stakeholders will provide advice on what may be in a programme theory, helping with refinement and sharing views on the credibility of a programme theory, along with suggestions as to where to find more data . They will be considered “content” experts as entitled by Wong (2018) and will be invited to be involved and asked to provide input at certain steps in the review as displayed in table 1 . It will be beneficial to share the
findings with health researchers and policymakers such as the National Institute for Health Research (NIHR) and Health Research Authority (HRA), but it is not possible for the scope of this review to involve them in discussions as this would require more time and resources to do so.
Due to the current social distancing requirements associated with the COVID-19 pandemic discussions with stakeholders will be held using the Microsoft Teams online platform unless stakeholders indicate a preference for other technology that would support accessibility. Stakeholders will be sent an invitation to join the meeting via EH, to limit accessibility to those invited. There will be two, separate stakeholder group meetings, one for health professionals/clinical academics and one for NIHR (Public) Research Champions, that will enable uninhibited discussions . A maximum of 10 participants per meeting will be set to provide the opportunity for all to speak. Stakeholders will be contacted via email before the scheduled meetings to offer a brief training session on Microsoft Teams (if required) and a summary of what to expect to ensure meetings run smoothly.
Patient and Public Involvement
The initial ideas for this review were discussed with NIHR (Public) Research Champions at a local, West Midlands primary care Patient Public Involvement (PPI) meeting held by EH. It was agreed that the topic is important and relevant to patients and the public. NIHR public Research Champions are volunteers who raise awareness about health research, in particular with those whom are less likely to participate . NIHR (Public) Research Champions will form a key stakeholder group and will be central to providing expertise at the second and third iterations of the review. At the end of the review, they will comment on the findings and contribute to the dissemination plan through helping to write a lay summary and designing a leaflet for dissemination to their peers.
Patient and Public Involvement through NIHR (Public) Research Champions will provide insights and opinion on the informed consent process including what it should look like in health research, along with sharing valuable input throughout the realist review steps . PPI will be facilitated through inviting an existing Equality, Diversity and Inclusion working group from the Clinical Research Network West Midlands that consists of NIHR (Public) Research Champions whom are interested in tackling health inequalities within health research specifically. In addition to the NIHR (Public) Research Champions, snowballing of interest from other PPI volunteers whom express interest in providing expertise will be invited at their request, to comment at planned meetings also.
In addition to the PPI stakeholder group, a multi-disciplinary group of health care professionals, clinical academics and the PPI Lead of the CRN West Midlands Equality, Diversity and Inclusion working group from the West Midlands will be invited for consultation, drawing on their expertise and insider knowledge related to the informed consent process in health research and under-served populations . The group collectively will have a variety of experiences including: carrying out the informed consent process with local populations from all across the West Midlands, including rural and urban areas and in various settings, such as care homes, secondary care settings and general practice; experience of recruiting participants from under-served populations; a vested “stake” in reducing health inequalities in health research and also insight into the difficulties of recruiting potential participants from under-served populations in health research. This experience and knowledge will be beneficial when prioritising, refining programme theory and building
CMOC’s (Context, Mechanism, Outcomes Configurations) . Some professionals may have experience of not being able to carry out the informed consent process with particular patients, or in certain locations and these experiences are also considered vital in helping to provide explanations of what works and what doesn’t, for whom and in what context.
As recommended by Pawson et al (2005), a series of steps in accordance with standard Cochrane headings can provide a helpful approach to carrying out a realist review. Therefore, this review will be conducted in this logical way .
Step 1. Scoping search and identification of a programme theory
It is recognised that it is natural for a scoping search to be an iterative process and often challenging, due to the complexity of the intervention under study . This may require refining of the research question(s) for a length of time during the review . EH will carry out the scoping search based on terms focussed on the intervention (informed consent). This will enable identification of existing theories about how under-served populations experience and understand information provided during the informed consent process in health research and in what context, and under what conditions it is effective, or ineffective. A combination of electronic databases, along with websites such as the Health Research Authority (HRA) will be searched, see table 2. It is also acknowledged that through discussions with stakeholders, further sources may be identified and used to inform the programme theory. The support of an Information Specialist to support searching will also be requested, as recommended by Pawson (2006) .
Type of Source
Name of source (s)
EMBASE, MEDLINE, Web of Science , PsychINFO
UCL IRIS ( Institutional Research Information Service) ,
HRA, MHRA, NIHR, (formerly INVOLVE), legislation.gov.uk
Search engines (Google), Editorials, Opinion pieces
e.g. reports, conference presentations
A list of important intervention theories for exploration will be compiled that will be of relevance to the research questions. Following the creation of a list of all relevant programme theories, they will be shortlisted according to which are deemed to be the most important for further exploration .
Step 2 Evidence searching
Purposive sampling will be used to search the literature. Using this sampling method will enable an iterative process that will allow repeated searching and refinement of search terms as understanding develops throughout the review . Pawson et al (2005) suggest approaching the search in four parts as follows:
- Carry out a background search of the literature
- Develop, refine, verify, or disprove a theory based on available evidence
- Implement the search strategy to identify relevant evidence
- Perform a final search that will help to identify further studies that may contribute to sharpening the programme theories discovered.
Free text and MeSH (medical subject headings) will be used to to ensure thoroughness. Each database will require an individual search which will be fine-tuned accordingly, to ensure relevant papers are identified. The reference lists of papers included will be examined for locating further papers that may add to the richness of data. To ensure validity and diligence of the search process experts within the field of interest will be contacted to verify results. Following each stage of the search, it will be essential to check whether any new literature adds anything different to the knowledge around the intervention (informed consent process). This will help to determine when saturation has been reached .
Searching will be conducted and reported in accordance to RAMESES standards . An example of an initial search strategy for MEDLINE, containing the search terms, is available in Additional file 1 (Search 1).
Step 3 Critical appraisal and data extraction
Preliminary screening of the literature using article titles and abstracts will identify all papers that are potentially relevant. Secondary screening of full-texts by the main reviewer (EH) will be carried out according to pre-determined inclusion/exclusion criteria (see table 3). A second reviewer will screen a 10% sample of the results, for agreement, with a record of exclusions made throughout the screening process. Any disagreements will be settled by the stakeholder group. Records will be managed using Endnote Online to manage references.
According to Wong (2018) realist reviews must take a logical approach in considering the trustworthiness of data . It is important to note that because this review is developing theory and data will come from many sources, Wong (2018) suggests assessing the trustworthiness of data by :
- Presuming that empirical data has been acquired methodically.
- Deciding if there is lack of clarity around data collection methods, in which case trustworthiness should be regarded with uncertainty.
- Making the effort to locate multiple, pertinent sources of data in relation to facets of programme theory. Relevance is key .
In addition to this, utilising a data extraction form will help to provide a reliable approach to data extraction . The data extraction form will be developed once a theoretical framework has been established as this will inform the questions that will need to be asked, to determine if the evidence from the theory areas should be included in the evidence synthesis. The data extraction form will be piloted on two articles before implementation, but an open-minded approach will be taken as it is understood that it may be necessary to have several forms to avoid a one-size-fits all approach to extraction .
Data extraction forms will be used in conjunction with Pawson et al’s (2005) recommended approach to gathering information through note-taking and annotation [31, 43-45]. Utilising these methods will provide a comprehensive and auditable trail for data extraction and critical appraisal.
Step 4. Organisation of the evidence
Synthesis of the evidence will aim to develop, test and refine the programme theory through analysis of the data . Data will be mapped to create a data matrix according to CMOC’s which will help with interpretations and in producing theory . Data synthesis will be guided by the approach used by Rivas et al (2019) to support interpretation and development of the programme theory. Following a similar approach will improve methodological quality through :
- Comparing evidence to gain better understanding
- Amalgamating data that provide evidence of corresponding mechanisms and outcomes
- Further reflecting on evidence that demonstrates opposing outcomes, but similar contexts
- Considering the circumstances of evidence that finds different contexts and outcomes
- Using judgement to determine methodological strengths and weaknesses [30, 47].
Step 5 Refinement and validation of the programme theory
The result of the realist review will provide government agencies, IRBs , researchers and health professionals with an explanation of what is effective, what is ineffective and what is unknown about the informed consent process for improving experience and understanding to aid decision-making in under-served populations. These recommendations and findings will include input from the identified key stakeholders and will form what the programme theory is. They may also contribute to further recommendations for developing training on how best to deliver the informed consent process and highlight potential avenues for exploring the design of health research studies.
Ethics and Dissemination
Ethical approval is not necessary for this study although all stakeholders will be fully informed of the study intentions. However, there are several ethical considerations to make around patient and public involvement as recommended by Pandya-Wood et al (2017) . Some of these ethical considerations will also be applied to the professional stakeholder group as to ensure a professional approach is taken. These include: role of the public contributor; avoidance of tokenism; the opportunity to withdraw; fairness of opportunity; taking a sensitive approach; respecting confidentiality and valuing public contributions  (see table 4).
Ethical considerations for Patient Public Involvement
Role of public contributors
The role of the public contributors will be made clear when they are invited to be part in the stakeholder group, through written information with the opportunity to discuss verbally if they wish. Their contributions will include developing a working definition of the term under-served, involvement in refining programme theory providing their ideas as to what is important when considering and refining programme theory, with sufficient time allocated to allow for discussion and flow of ideas.
Avoidance of tokenism
Details of PPI contributions and how they will be utilised and shaped programme theory will be described in the review.
Opportunity to withdraw
It will be clearly communicated to public contributors that they may withdraw their involvement at any stage in the review process without needing to provide a reason.
Fairness of opportunity
The researcher will seek to engage pragmatically, with an already formed group that represents equality, diversity and inclusion. This will also be a limitation, because the scope of this review will not allow more time, or funds to engage with a larger group.
A sensitive approach will be taken with the public contributors with every effort to ensure personal information is not revealed in relation to individuals that are involved, whilst being aware of any potentially, undesirable reactions of members of the group.
It will be clearly stated to public contributors that should any personal issues be discussed during the meetings, that this information will remain confidential.
Valuing public contributions
A pragmatic approach will be taken with a view to being flexible with technology, utilising what works best. Regular communication to all public contributors will be carried out with their involvement described within the review and how it was included.
The expertise from NIHR (Public) Research Champions, or public contributors, during stakeholder meetings will help to identify the concepts that will build CMOC’s, through making judgements about text, whilst utilising the data to confirm, refute, or refine aspects of the programme theory Wong (2018 ).
Findings from the review will be disseminated to patients and the public (in the form of a lay summary), researchers, health professionals and policymakers in relation to the informed consent process with under-served populations. The findings will offer deeper understanding about the relationships between the informed consent process and the contexts and mechanisms which influence decision-making by under-served populations. Findings will be shared as set out in table 5. The aim of disseminating findings with policymakers is to influence their thinking .
Patients and the public
Centre for Engagement and Dissemination (CEDA) (lay summary)
Public Involvement and Lay Accountability in Research and innovation PILAR) (lay summary)
Publication in a peer reviewed journal
Summary of findings emailed to HRA Head of Policy
West Midlands Research Champion Forum (presentation)
Poster at academic conference
Summary via NIHR Signals and Connect newsletters
NIHR Patient and Public Involvement and Engagement (PPIE) Google Community (link to lay summary)
Poster Presentation at West Midlands R&D Forum
Email MHRA Director of Inspection, Enforcement and Standards with summary
Twitter (lay summary)
Twitter/ Warwick Medical School Unit of Academic Primary Care webpage ( summary of findings)
Twitter (@HRA News, @MHRApress) - link to findings