Individuals with intellectual disability

Individuals with ID often present with communication and cognitive functioning challenges, which may make obtaining informed consent more problematical [4]. An added layer of complexity for people with ID, is that they are likely to have co-morbidities which may increase their physical vulnerability, and, with a lack of insight into the prevalence of health conditions in this population [10–11], access may be confined to smaller numbers of eligible participants [12].

Researchers often experience more complex engagements with family members and National Health Service (NHS) health and social care teams when including people with ID in research studies [3]. As a result, decisions about inclusion of these individuals may be made by multiple people at multiple time points, for example, by NHS health and social care staff or by study team members (when considering screening and eligibility criteria), or by other individuals with a caring role making paternalistic decisions based on their perception of the potential trial participant’s competence/willingness to participate.

RCT methodology

There are practical issues around accessible research design and adapting resources, such as the participant information sheet and consent form [13], and the need for multiple consent procedures [14], which may lead RCT researchers to intentionally or unintentionally exclude people with ID from their studies [15]. Such exclusion may deny this group the benefits from improvements in treatment which will be subsequently available to the general population [16].

In recognition of the challenges around recruitment for specific populations [17–18], tools such as PRECIS-2 have been generated to help researchers with their design decisions, to give greater focus and support to addressing such issues [19]. However, despite growing recognition of the complexity related to recruitment into RCTs, and particularly for specific populations, there remains a lack of effective solutions [20]. While these issues have been raised before, how they influence the decision-making of people responsible for identifying and recruiting people with ID has rarely been considered [21].

NHS health and social care systems

The complexity of including people with ID in RCTs, as described above, can make it difficult for researchers to secure adequate funding to carry out trials, despite the development of the UK Research Framework by the Health Research Authority (HRA) [5] which supports inclusivity. It can also be challenging to get buy-in from NHS health and social care organisations across the UK who have limited workforce resources to support individuals with ID, adding another layer of complexity to accessing this population [22]. The involvement of disparate organisations across health, social care and academia inevitably leads to a certain amount of bureaucracy and can result in delays within research governance, finance, and staff recruitment processes prior to the commencement of the identification and recruitment of potential participants [23].

An added complication has been the impact of the COVID-19 pandemic on healthcare in general and on the ability of organisations to prioritise and support research currently [24]. The impact of limited routine healthcare for people with ID during the pandemic has been identified [25], however, the current impetus to address significant backlogs in healthcare delivery for the public, coupled with the aforementioned challenges conducting RCTs with people with ID, suggests that anecdotally, research priorities are less likely to focus on this smaller cohort.

Type 2 Diabetes (T2D) is reported to be 2–3 times higher in adults with ID than in the general population [26–27], due to certain chromosomal conditions (i.e. Down’s Syndrome) increasing the risk of developing T2D. People with ID are also more likely to lead unhealthier lifestyles that include poor diets, decreased physical activity, increased sedentary behaviour and greater use of antipsychotic medication leading to higher levels of abdominal obesity [28]. Furthermore, people with ID develop T2D at an earlier age making management protracted and more complex [29].

Previous studies have identified initiatives designed to promote healthier outcomes for people with ID/T2D [29–30] and the current study is an RCT using a specifically designed programme for this population.

The my diabetes and me study exemplar

The structured diabetes self-management education programme – DESMOND runs successfully via the NHS in the UK and is available to the general population who have been diagnosed with T2D [31]. It has not, however, been accessible to people with ID/T2D [11,26]. An earlier pilot feasibility study demonstrated that a modified structured self-management education programme known as DESMOND-ID was acceptable and can be successfully delivered to adults with ID/T2D [32]. The National Institute for Health and Care Research (NIHR) has recently funded a multi-centre RCT with an internal pilot to test the efficacy and impact of the DESMOND-ID intervention in study sites across the UK (My Diabetes and Me Study – HTA NIHR 131692). Three sites participated in the internal pilot study with a further eight sites participating in the full study. Participants were randomised to the Intervention Group (DESMOND-ID Structured Education Programme) or the Control Group (education booklet and treatment as usual). The earlier pilot feasibility study identified that the identification and recruitment of adults with ID/T2D was more challenging than anticipated [29]. With this in mind, an established intervention – the QuinteT Recruitment Intervention (QRI) – was embedded within the internal pilot of the NIHR definitive study to focus on and address the complexities and barriers of recruiting adults with ID. The aim of this QRI study was to understand and address the enablers and barriers in how clinical staff identified and approached adults with ID/T2D to participate in an RCT. In this paper, we report on those enablers and barriers using the ‘My Diabetes and Me’ Study as an exemplar.

The QuinteT Recruitment Intervention within the internal pilot of the ‘My Diabetes and Me’ Study

COREQ (COnsolidated criteria for REporting Qualitative research) Checklist is included in S1 File.

The purpose of the QRI was to identify and address issues that were hindering effective recruitment [33]. Table 1 illustrates the QRI – supported triangulation of multiple data sources including:

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Table 1. Data collection methods.

https://doi.org/10.1371/journal.pone.0332744.t001

• interviews with key stakeholders.
• audio-recording of recruitment-to-study discussions.
• detailed screening logs using the SEAR framework [34].
• mapped recruitment pathway.
• review of study documentation (easy-read Participant Information Sheets (PIS) and invitation letters) and minutes of Trial Management Group and Investigator meetings.

Recruitment of staff began on 21^st October 2022, with data collection beginning on 4 November 2022. Given the delays in appointing staff into post which will be discussed, recruitment and data collection of staff continued until 29^th September 2023. Staff were identified through their involvement in the RCT and a snowballing process via management and research teams. Recruitment of people with ID began on 1^st April 2023 with data collected from 26^th April – 15^th August 2023. Individuals were contacted when they had been randomised to the study. These data were used to investigate how the study recruitment process worked (enablers) in each study site, and to elucidate key bottlenecks (barriers). Findings were synthesized and discussed with the study’s Chief Investigator and trial management group (TMG), and actions formulated and implemented to optimise recruitment in the pilot stage, or to offer recommendations for the full RCT.

Semi-structured interviews were undertaken by the first author (RK) with NHS health and social care staff involved in the identification of potential participants with ID/T2D, either through their professional role, or as leaders within NHS services across three sites in the UK. These included community ID nursing teams, diabetes nurse specialists, GPs, and practice nurses. RK is a registered nurse with a PhD. This author was the lone data collector with additional responsibility for data curation, development and refinement of aspects of the documentation as it applied to this work, e.g., screening logs, topic guides etc., project administration, transcription and coding in partnership with other authors and writing all manuscript drafts.

Research Associates and Clinical Research Nurses (CRNs) employed for recruitment and data collection duties along with senior leaders employed in clinical research networks related to the UK sites were also interviewed in February/March 2023 to provide insight into some of the organisational challenges related to multi-site research that had been identified in earlier interviews. People with ID/T2D who had consented to randomisation within the internal pilot were also invited to take part in an interview with the first author (RK) to explore their views on the study and the recruitment process. Topic guides were developed drawing upon findings from the feasibility study [32] and previous QRI studies [35]. These were revised as data collection and analysis progressed to advance developing themes from earlier interviews as detailed in S2 and S3 Files.

Participants were not asked to review their transcripts. A meeting was held after the initial data analysis had been completed, between the research team and many of the professionals interviewed. Early findings were discussed, and further insight was added to the data collection to enhance the coding process (Fig 1). People with ID who took part in the interviews were not sent their transcripts for review, but an easy-read summary has been produced as part of the dissemination process.

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Fig 1. Coding tree.

https://doi.org/10.1371/journal.pone.0332744.g001

Data analysis

Data from the interviews and recruitment discussions were subjected to full transcription and de-identification by the first author (RK). All transcriptions were pseudoanonymised using a description of role/site/interview order. Interviews with staff lasted 35–50 minutes while interviews with people with ID/T2D lasted 9–20 minutes. A code book detailed in S4 File was developed based on both inductive and deductive thematic analysis which was regularly shared and discussed with the team [36]. A theoretical framework was not used for this study as the team wished to approach the study with an open mind and with equipoise, looking at the data independently and collectively, with regular discussions as codes were added to and refined as more data were collected. This is in keeping with the pragmatic approach adopted during the initial development of the QRI approach, which used multiple methodological underpinnings [33].

Further independent analysis on a selection of interview transcriptions was carried out by VC, and recruitment discussions were analysed independently by NM and discussed amongst the research team to ensure trustworthiness of the process. Documentation analysis by the first author (RK) included screening logs, recruitment pathways at each site, and agreed minutes of all meetings. Issues arising from the documentation were added to the existing codes, with new codes added as needed. The findings relating to aspects of recruitment which appeared to be working well and those areas working less well were shared with sites to raise awareness and so that by working together, challenges could be addressed for the internal pilot and main RCT. Our experience of using the QRI methodology will be reported in a separate paper.

Ethics approval

The QRI was integrated after ethical approval for the internal pilot stage of the RCT had already been granted. Separate UK Research Ethics Committee approval was therefore sought and approved for the QRI component (22/HSC/0009).

Informed consent was obtained from all participants. Health and Social Care and research staff were provided with a Participant Information Sheet (PIS) detailing the study and a consent form which they were asked to sign and either return electronically to RK or sign on the day if the interview was face to face. Information about the QRI study was included in the easy-read format PIS provided to people with ID/T2D about the internal pilot study. The QRI interview was included in the recruitment discussion about the internal pilot study and written consent obtained. RK confirmed their continuing consent when the participants were contacted to arrange the interview, and again on the day of the interview.

Data sources

• Interviewees detailed in Table 2 included:
  • NHS health and social care staff including senior managers, clinical network staff and ID team members (n = 14)
  • Research staff (n= 6)
  • People with ID/T2D who were recruited to the study (n=5)
• Analysis of:
  • Recruitment discussions (n=6 from 1 site)
  • Screening logs from 1 site (n=20)
  • Minutes of study meetings (n=27)
  • Activity log of communication involved in coordinating the internal pilot and arranging the interviews (n = 1).

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Table 2. Interview characteristics.

https://doi.org/10.1371/journal.pone.0332744.t002

Thematic analysis of the core recruitment challenges

Fig 1 illustrates a coding tree was developed as art of the analysis process to illustrate the initial theme development and subsequent revisions until the final themes were agreed. The thematic analysis identified a number of themes which were aggregated into three core recruitment challenges: (i) study buy-in and engagement; (ii) discomfort putting potential participants with ID/T2D forward for the RCT; and (iii) the emotional labour arising from organisational challenges with RCT set up and conduct.

Interview data was the main source of evidence used to develop the themes, with other sources of data such as screening logs and minutes of meetings examined to support what was being said in the interviews or to offer additional insights into the management of issues as they arose. Only one NHS site returned screening log evidence illustrating that although a large proportion of patients were screened for eligibility (n = 187) and 142 were deemed eligible, only 65 were approached so they could consider taking part and 39 were subsequently randomized (unsuitable n = 13; declined n = 13). Data from this log, and interviews with NHS health and social care staff from the other two sites indicated that most people were excluded early in the recruitment pathway as they were considered not suitable for the study (even if they met the eligibility criteria as per protocol) or they, or their carer, did not wish to meet to hear more about the study. Those that were happy to meet with a researcher to discuss the study tended to consent to participation (75%).

Theme 1: Study buy-in and engagement

Complexities of engaging multi-specialty teams.

Researchers in the study had to work closely with different ID organisations to identify potential participants given that services for people with ID and T2D can be accessed through multiple pathways in the community, as shown in Fig 2.

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Fig 2. Recruitment pathway and the multidisciplinary teams involved.

https://doi.org/10.1371/journal.pone.0332744.g002

While all the NHS sites identified multiple teams who could assist with identification and recruitment, for example supported living and residential facilities, day centres, and voluntary support organisations, community ID teams were considered to be the richest source to identify potential study participants, given their expertise in working with people with ID. By starting study preparation in advance of initiating the recruitment process, Site 1, who reached their recruitment target and recruited the majority of the total number of consented adults with ID/T2D in the internal pilot, had community ID teams engaged with initial screening, and the research team reported an effective collaborative relationship with them. As the study progressed, it was noticeable that NHS sites 2 and 3 who were less successful in terms of recruitment, had less time for study preparation due to bureaucracy delays, resulting in struggles to engage community ID teams in the screening process and those who used clinical research network facilities to conduct the recruitment discussions. It was also noted that screening by other services at site 1 and reports at meetings from sites 2 and 3 illustrated that staff with less experience of working with this ID population were more inclined to exclude greater numbers of potential participants at either the initial screening stage or at the recruitment discussion stage, and had a less successful collaborative working relationship with the site research team:

“But whenever it comes down to it, I’ve been chasing, chasing, chasing, you know, promise me, you’re gonna generate a list of potential participants. And that’s been quite a struggle because it’s like, follow up telephone conversations, phone calls, which have been returned back to me follow up emails, follow up, but nothing has come from us, which is disappointing given the initial meeting.” (Researcher 03002)

What was underestimated as the pilot study progressed was how much engagement the research team needed to have with NHS health and social care teams to reinforce the potential benefits of the study, before researchers ever got to speak to potential participants with ID. It became evident that fostering a strong relationship between NHS health and social care ID teams and research teams was required to enable a good understanding of the study and its rationale, and to support buy-in:

“…So, whenever you do research it’s really, really important that you have a very strong relationship with your clinical partners way before the intervention starts and get their buy-in. So they would have ownership as well of the project. I think it’s really difficult just to rock up to someone and say ‘hey, do you want to be involved in a trial?’ Understanding the trial, understanding all the processes has been very, very complicated.” (Researcher 01007)

Theme 2: Discomfort putting potential participants with ID forward for the RCT

Theme 3: Emotional labour arising from organisational challenges

Where do the challenges lie?

Many individuals with ID/T2D, once identified and approached, were open to the study and willing to consent, and this was an ongoing source of motivation and perseverance for all the site teams. Challenges were more prevalent in NHS site set up and the early stages of the identification and recruitment process, in terms of identifying and approaching potential participants with ID/T2D, as opposed to the later stages of gaining participant consent. Issues were identified with setting up and conducting an RCT with people with ID in a post COVID-19 climate, getting senior and front-line staff buy-in and engagement with a study that supports self-management of health for people with ID, and discomfort putting people with ID forward if staff felt they had limited experience with or had preconceptions of an ID population. As these barriers to recruitment increased, at times seeming to be intractable, the emotional burdens for the research team charged to deliver the study on time and in budget mounted.

Clear obstacles and hidden challenges

While using the QRI is a novel approach within ID research, many of the issues identified will resonate with researchers in studies outside of the ID world [44]. In some ways, this is reassuring as it illustrates that the challenges of recruitment into RCTs cannot be assumed to solely relate to this ID population itself, which is a common misconception. What the QRI has enabled, however, is the identification of recruitment challenges that were clear to those involved – ‘clear obstacles’ [45] – namely the organisational and logistical issues to do with staff recruitment and opening delays. But it has also unearthed the ‘hidden challenges’ [43,45,46] of recruitment such as making judgements on who would be suitable for the study based on preconceptions, which equally hampered recruitment but in a way that was perhaps less obvious to those recruiting. The number of NHS health and social care staff/family and paid carers making judgements on behalf of the ID population is a consistent theme relating to recruitment challenges in research [47–48]. This is an alarming indication of how health and social care professionals are failing to support this ID population to engage in clinical research that could benefit their health [49]. When viewed from an advocacy perspective, it is crucial that people within this population are not taken advantage of. However, from a paternalistic point of view, it excludes people with ID who may be able to make a valuable contribution to research and, demonstrated through their interviews in this study, their pleasure at being included.

Gatekeeping

There is a significant cohort of NHS health and social care staff, and paid carers, referred to as ‘gatekeepers’ who have contact with people with ID, which would not be the case in clinical research with less vulnerable populations [50–51]. This creates an additional burden on researchers in relation to communication strategies and emotional labour [3,52,53]. NHS health and social care staff and paid carers acting as gatekeepers can be seen as a protective enabler for the ID community when it comes to ensuring that the ethical standards around capacity and participant wishes are maintained (50), but several layers of gatekeeping can present a systemic barrier that researchers struggle with [48]. Recruiters may have to contend with the intellectual challenge of presenting the study to an individual who fulfills the eligibility criteria, but in whom they feel would be better off with one intervention over another [46,52]. This has the potential to compromise equipoise in their discussion of the study and influence the potential participant’s decision making, but can be overcome with appropriate training and support.

The frustration of organisational barriers

In this study it wasn’t so much about how the study was presented to potential participants with ID/T2D or those individuals declining participation due to intervention preferences, but issues much earlier on in the identification and recruitment process. Many of the challenges arose because of insufficient preparation, readiness, and lack of early involvement of the NHS health and social care senior managers and front-line staff. As a result, the work required behind the scenes in this study became increasingly apparent and created an emotional burden. The emotional aspects of working on a clinical trial for those undertaking recruitment has been reported previously [45,47,53], but less so from the angle of those involved with the overall design and delivery, as in the present study. Communication with participants in any research study is important, but it was clear from these interviews that early and ongoing engagement with both NHS health and social care staff and adults with ID/T2D and their families/paid carers is a core key component to timely and effective RCT recruitment.

Preparing an RCT submission

There are a number of challenges researchers need to be cognisant of when preparing RCT grant submissions related to any population. Realism and pragmatism must play a big part in any research study, and this should begin with the grant application. There is a tendency to underestimate the budget required for clinical trials and many researchers seek to minimise their request in the hope of a successful grant application [54]. It is also common for only 80% of funding requested to be provided, which may further hamper recruitment efforts later. Research may not be a current priority within NHS health and social care, given the issue of backlogs in care provision partly as a result of the COVID pandemic, which may impact a study’s ability to recruit [55] as noted in the present study. As a result, realistic funding must be included to ensure that sufficient high calibre staff are seconded/employed to prioritise research studies, particularly in those populations where additional time is required for recruitment processes, such as the elderly, children, and people with cognitive, intellectual and developmental disabilities. It is also crucial to provide realistic deadlines for study procedures including identification and recruitment. These are often optimistically tight, leading to anxiety and potential conflict when they are not adhered to. Early discussions with study sites to confirm what is achievable are important and recording of decision-making at each stage supports accountability throughout the study.

Leadership

Support and leadership at every level is essential for a successful trial [56]. This can be difficult to achieve unless researchers engage with NHS organisational leaders at an early stage. Prioritising a research agenda within NHS healthcare has always been challenging [57] and involvement of the relevant organisations in developing the grant submission is one way of ensuring ‘buy-in’ at an early stage, but researchers should be cautious of assuming that this alsomeans ‘buy-in’ from NHS health and social care staff, which may not be the case [23]. Early andregular conversations with NHS senior managers and those front-line staff responsible for recruitment are central to the success of any trial. A positive relationship between NHS health and social care staff and research teams can dictate the success of a recruitment strategy, providing clarity of each other’s roles and a cohesive approach to recruitment [58]. As we have seen from this study, building, and maintaining relationships is crucial but problematic in NHS health and social care as capacity and capability are increasingly mentioned as barriers to recruitment [59]. This can be an issue for any study, particularly those involving multiple specialties, perceived capability for vulnerable populations or even patient ownership [46,48]. Pre-recruitment work should therefore focus on establishing or renewing those relationships with senior NHS leaders and health and social care staff at each site to address the other obstacles that are an inevitable part of any research study.

Strengths and limitations

This is the first time the QRI has been incorporated into an RCT involving an ID population with associated health co-morbidities such as T2D. This allowed us to triangulate multiple sources of data to understand how the identification and recruitment processes worked in real-time and where the key bottlenecks were [33]. Although identified issues such as challenges with setting aside personal views when considering study eligibility have been noted previously [52], embedding the QRI in a trial with an ID population enabled us to understand how this specific context shaped previously reported issues, which will be of value to future ID trialists. The QRI was also able to develop and implement actions to address the issues identified, either as the pilot study was underway or to implement in the full study to maximise successful recruitment in this and future trials.

A limitation of this work is the lack of screening log information supplied by two of the study NHS sites which would have helped identify clearer decision-making pathways and unpick any site-specific bottlenecks. Only Site 1, who randomised the majority of participants to the study, provided screening log data. A significant amount of work went into developing the screening logs, but clearly the lack of engagement in returning the information needs to be explored for future studies. We also recognise that we could have included more interviews with participants with ID/T2D who were recruited to the study and perhaps some family carers to provide further insight into the recruitment approach from their perspective. We hope to address this with a further piece of work to capture the views of family/paid carers to strengthen the overall findings from the study. However, we do recognise that the recruitment challenges lay less with the individuals with ID and more with NHS organisational and team working aspects, so it is unlikely that further data from participants themselves would have changed the narrative presented.

Recommendations

On the basis of findings from the present study, the following implementation recommendations are made to optimise recruitment of adults with ID to randomised trials that support better health:

• Invest time with NHS senior managers and front-line health and social care study site teams in the early stages of study set-up to:
  • establish relationships, expose views on the study such as selection/gatekeeper bias
  • identify training needs for ID nurses such as familiarisation with research processes,
  • promote study buy-in
  • explore anticipated challenges such as identifying how/where potential participants can be approached, e.g., community groups, day centres etc.
  • ensure ongoing monitoring of these issues as recruitment is underway.
• Support NHS sites to collect detailed data on screening and recruitment processes to identify where the core recruitment barriers are and whether they are study wide or site specific. Site staff should be aware of the value of collecting such data and results fed back and discussed to raise awareness and resolve identified issues.
• Highlight specific training issues for CRNs working with the population under study to address potential discomforts such as communication skills on explaining research studies to this population.
• Adults with ID/T2D are generally willing to participate in RCTs to improve their health but have concerns that could act as barriers. Recruiting staff should be supported to elicit and explore their intervention preferences and concerns to support informed decision making on study participation.
• Acknowledge the pressure on the grant holders and particularly the CI, to deliver a funded study on time and in budget despite a plethora of obstacles and barriers that may be beyond their control.
• Advocate for a review of training and support for PIs and co-investigators re roles and responsibilities and problem solving of recruitment challenges.