Participant experience of invasive research in adults with intellectual disability

Abstract

Clinical research is a necessity if effective and safe treatments are to be developed. However, this may well include the need for research that is best described as 'invasive' in that it may be associated with some discomfort or inconvenience. Limitations in the undertaking of invasive research involving people with intellectual disabilities (ID) are perhaps related to anxieties within the academic community and among ethics committees; however, the consequence of this neglect is that innovative treatments specific to people with ID may not be developed. Such concerns are likely to continue while there is limited published knowledge regarding the actual experiences of people with ID who have participated in invasive clinical research. As part of a pilot study trialling the novel use of a surgically inserted device to curb overeating in people with Prader-Willi syndrome (PWS) we have investigated the experience of research through semistructured qualitative interviews involving three participants and their carers. Thematic analysis revealed that the adults with PWS and their family carers rated their participation positively, seeing it as a rewarding and enriching experience. This brief report discusses findings from our interview data in order to highlight strategies which may ensure that research is acceptable to participants, meets the necessary ethical standards and is able to achieve the aims set out by the researchers. To our knowledge, this is the first study to record experiences directly from people with PWS and their carers regarding their involvement in invasive clinical research.

Designing genetic studies for people with intellectual disabilities: Practical lessons from a pilot study

“…Although research participation was paired with clinically indicated blood monitoring, many residential home staff declined to facilitate a Best Interests model of substitute decision‐making in the absence of an Independent Mental Capacity Advocate (IMCA). Unwillingness or inability of support workers to facilitate participation of PwID in research involving invasive procedures is common and our study confirms it (McAllister et al, 2013).…”

From Tokenism To Self-Determination: Ethical Considerations in Research with People with Intellectual Disabilities

“…John et al, 2022); d) Inaccessible language and documents(St. John et al, 2022); e) Use of offensive or outdated language in research policies and associated documents(St. John et al, 2022); f) Difficulty in recruiting diverse people among the group, which includes the possibility of coercion from caregivers, family members and/or legal guardians(Goldsmith & Skirton, 2015;St. John et al, 2022;Tuffrey-Wijne et al, 2008); g) Difficulty in obtaining consent(McAllister et al, 2013;St. John et al, 2022); h) Systematic exclusion related to participation(St. John et al, 2022); i) Protection of ethics boards(McAllister et al, 2013;Watchman et al, 2019); j) A high probability that it will be necessary to request an extension of time, which implies a restriction on costs(McAllister et al, 2013); k) Possibility of gatekeeping by associations/organizations, caregivers, family members and legal guardians(Tuffrey-Wijne et al, 2008); l) Lack of privacy that PwID may be subject to from caregivers(Hall, 2013); m) Lack of knowledge, on the part of ethics councils, about capabilities, needs, correct "(...) the form itself [ for ethical approval] is a barrier to inclusion (..…”

Do Tokenismo à Autodeterminação: Considerações Éticas na Investigação com Pessoas com Deficiência Intelectual

“…; g) Dificuldades em obter consentimento(McAllister et al, 2013;St. John et al, 2022); h) Exclusão sistemática relacionada com a participação(St. John et al, 2022);i) Proteção dos conselhos de ética (McAllister et al, 2013; Watchman et al, 2019); j) Grande probabilidade de ser necessário pedir uma extensão de tempo, o que implica uma restrição nos custos (McAllister et al, 2013); k) Possibilidade de haver gatekeeping por parte das associações/organizações, de cuidadores, familiares e responsáveis legais (Tuffrey-Wijne et al, 2008);…”