Barriers

Despite the amount of research conducted over time, many challenges that have been reported previously still remain and have an impact on the completion of studies. Lennox et al^{Reference Lennox, Taylor, Rey-Conde, Bain, Purdie and Boyle4} described identification of substantial numbers of participants, frequent need for substituted decision-making, occasional limited literacy of both person and carer, and organisational gate-keeping practices as significant barriers to recruitment in an RCT of health checks in Australia. More recently, a systematic review of 53 papers reporting RCTs in people with intellectual disabilities published between 2000 and 2017^{Reference Mulhall, Taggart, Coates, McAloon and Hassiotis5} identified similar barriers in recruiting to target, participant treatment preferences, engaging with stakeholders, obtaining consent and staff turn-over. Optimistically the authors concluded, ‘conducting RCTs with people with cognitive disabilities can be challenging, however, with reasonable adjustments, many of these barriers can be overcome’.

People with intellectual disabilities are excluded from research that may be relevant to their health vulnerabilities,^{Reference Spaul, Hudson, Harvey, Macdonald and Perez2} as well as being sceptical about the impact of it on their lives. This is illustrated by the Research Voices project,⁶ which revealed a number of serious concerns that parents of people with profound and multiple disabilities harbour about research, such as mistrust of health professionals seeking participants for studies, the emotional and time burden of research assessments, frustration with not knowing the findings or findings not translating to real improvements in practice.

Research infrastructure brings its own multifaceted challenges. The role of clinical research practitioners (CRPs) (who are National Health Service (NHS) based and able to recruit from services directly) is not fully understood by intellectual disability services and there is significant variation among the operations of clinical research networks across the different UK countries. Therefore, professionals in the services may be asked to undertake recruitment in addition to an already busy clinical role. Further, data guarantors are frequently local authorities, who are providers of social care services not directly connected to the NHS in England. Finally, intellectual disability services that are located outside NHS structures have fewer opportunities to be informed of ever-evolving research processes, thus remaining unable to utilise available resources to assist them in incorporating and supporting research in their day-to-day practice.

Clinician factors are also important in maintaining non-engagement in research activities, including older age, being male and working in the private sector.^{Reference Rye, Friborg and Skre7} Oliver-Africano et al^{Reference Oliver-Africano, Dickens, Ahmed, Bouras, Cooray and Deb8} identified beliefs about drug efficacy, potential ethical conflicts in medication trials and multidisciplinary team processes as having adversely affected recruitment to a clinical trial of antipsychotics in adults with intellectual disabilities. Delays associated with any of these factors inevitably matter in completing studies that depend on time-sensitive research contracts and are likely to hamper the validity of the research findings if there is underrecruiting.

Finally, clinicians and scientists in general may not make sufficient effort to include participants from underrepresented groups in their studies, thus perpetuating the limited access of people with intellectual disabilities in research. This is particularly important for diseases where it has been demonstrated that there is excess morbidity and/or mortality in this population.⁹

Facilitators

Prioritisation of research is likely to confer benefits to both health and care organisations, as shown by views reported in a review of engagement in research:^{Reference Hanney, Boaz, Jones and Soper10}

These lessons, although not specific to intellectual disabilities, are relevant in this context as presenting a justification for embracing research by the multitude of service configurations delivering care to this population.

In other changes to research governance, the new Health Research Authority has halved the time needed to obtain regulatory approvals,^{Reference Ranieri, McKay, Stynes and Kennedy11} although other milestones along the research pathway remain areas of concern.

Although the funding for research in intellectual disabilities may be lower than what is essential for investigating the increased morbidity, mortality and the health inequalities seen in this population group, there is an emerging cohort of completed high-quality studies. There are also many committed researchers, healthcare service professionals and other staff whose enthusiasm and problem-solving capacity signal their willingness to engage with the process. These may further promote interest in research by influencing national clinical practice and through targeted dissemination, including to people with intellectual disabilities and their carers. Clinician familiarity with academic work, peer support and support from management are also likely to increase positive attitudes towards research. Research that is seen as arising out of patient concerns and that could lead to tangible benefits in interventions and care improvements is also likely to be supported.⁶

Recently the NIHR Dissemination Centre published a themed review on intellectual disability research.¹² More than showcasing the funded projects, it highlighted the meaning of the findings for the care that people with intellectual disabilities and their family carers receive. In addition, the collection of studies included in the review demonstrate that funded research can be conducted successfully in the field of intellectual disabilities but that all the studies have had significant involvement of people with intellectual disabilities and their family carers throughout. These studies are examples of good practice that can be shared between researchers, people with intellectual disabilities, their carers and charities supporting them.

Solutions

A primary area for mitigation lies in health and social care professionals’ and people with intellectual disabilities’ conviction of the importance of research and its wider contribution to lives and well-being. A recent course^{Reference Tuffrey-Wijne, Kar Kei Lam, Marsden, Conway, Harris and Jeffrey13} devised to train people with intellectual disabilities in research methods suggests that learning about conducting research and driving the research process is feasible. Such courses could increase the number of suitably trained people with intellectual disabilities who could be recruited to work as researchers in various projects.

Incentives for encouraging donation of time to research activities by family and paid carers may improve uptake and retention, alongside other strategies. Increasingly, experts by experience are being asked to interpret and comment on research findings and this is a way of increasing familiarity with research processes, as well as consumer feedback.

The well-intended efforts of paid carers to protect vulnerable adults if they lack capacity, including the personal data protection regulations, often stifle participant recruitment. In England and Wales, this could be addressed by a revision of the Mental Capacity Act 2005. Heywood et al^{Reference Heywood, Ryan, Killet, Langdon, Plenderleith and Shiggins14} outlined how the Act is predominantly focuses on treatment and decision-making within a ‘best interests’ framework, rather than on research where decisions are not made using that framework. The sections of the Act governing research do not effectively balance protection and empowerment, and researchers may be reluctant to include participants who lack capacity in research projects.

The research community may also need to take some responsibility in providing solutions to the present challenges. Being clear about what the findings mean to the wider group of people with intellectual disabilities, proactive dissemination strategies and other activities in engaging the public with research are paramount in moving forward. Research aims to shape service delivery and to translate advances in science into measurable benefits for the population at large. It is questionable whether consumers consider research findings relevant to their health and whether those who commission services apply research findings to enhance clinical effectiveness and value for money.

An example of fostering closer links between clinicians, academics and people with lived experience of intellectual disability is the newly formed RADiANT consortium, a platform that works to increase health and social care staff's awareness of research and develop research skills and capacity. The consortium is focused on mental health and behavioural problems in intellectual disabilities, autism and other neurodevelopmental conditions (see radiant.nhs.uk for more information). So far it has produced guidance on how to manage the COVID-19 pandemic in different mental health settings and has delivered several educational activities. Its wider impact remains to be established.

Professional bodies across all professions must also promote research-related objectives in training curricula and on public-facing forums such as websites and newsletters.

Strengthening health and social care links is an area for further development, especially as social care is identified by NIHR as a domain for research investment. This means extension of the research infrastructure to reach the neglected care sector, which is central to accessing individuals to take part in research activities as proxy informants.

The coronavirus pandemic has shown that, while continuing to endorse ethically conducted research, it is possible to do so at pace.^{Reference Wise and Coombes15} It will be important to remember those lessons as we are coming out of the pandemic and in the event of future public health emergencies. In particular, they can inform how to carry out remote research assessments and interviews with participants with intellectual disabilities and ensure that the voice of experts by experience remains central to research activity. During the pandemic, people with intellectual disabilities have been disproportionately affected in both their health and social care needs and require high standards of support in both. We must be able to reassure them and their families that being partners in research pays off in achieving those standards.