Intellectual Disability Research Research Articles

Disseminating our knowledge in an inclusive society: An inaugural editorial statement

Inclusive research is essential at all stages of research, including data collection and dissemination. This essay investigates how current literature and the practical application of inclusive practices in the academic setting illuminate this critical but often overlooked aspect of research. It offers the South African policy of Working Paper 6 (WP6) to clarify how the principle and policy of education for all learners can be implemented in inclusive societies and development, which confront various obstacles due to psychosocial issues. It discourses the purpose of the South African Policy of WP6 and its discourses in multiple contexts by adopting innovative and more inclusive techniques for dissemination, intellectual and developmental disability research, and analysis that can reach far beyond the academic walls. It finally discusses barriers to inclusive dissemination and methods to overcome them. Even though further research needs to be explored in this regard, this article would be beneficial to scholars to champion and implement inclusivity to make research more meaningful and accessible to all who are affected by any form of exclusion and discrimination in our society.

The All Together Group: Co-Designing a Toolkit of Approaches and Resources for End-of-Life Care Planning With People With Intellectual Disabilities in Social Care Settings.

Support staff within social care settings have expressed a need for resources to facilitate end-of-life care planning with people with intellectual disabilities. This study aimed to co-design a preliminary toolkit of end-of-life care planning approaches and resources that can be implemented in adult social care services for people with intellectual disabilities. An adapted Experience-Based Co-Design process was applied to develop a toolkit for end-of-life care planning with people with intellectual disabilities. A co-design group (the 'All Together Group') met six times from January to October 2023. The group comprised nine people with intellectual disabilities (including four researchers with intellectual disabilities, who also co-facilitated the workshops), five family members, five intellectual disability support staff, two intellectual disability service managers,and five healthcare professionals. The All Together Group tested resources for and approaches to end-of-life care planning with people with intellectual disabilities, based on findings from a scoping review and a focus group study. Easy-read end-of-life care planning forms were deemed overwhelming and complicated, whilst visual and creative approaches were welcomed. Three new visual resources to support illness planning and funeral planning with people with intellectual disabilities were developed: (i) 'When I'm ill' thinking cards; (ii) 'Let's Talk About Funerals' conversation-starter pictures; and (iii) 'My funeral' planning cards. These three resources, alongside three positively evaluated existing resources, were included in a new toolkit for end-of-life care planning with people with intellectual disabilities. Through an iterative, flexible, inclusive,and comprehensive co-design process, a toolkit of three newly developed and three existing resources was created to facilitate support staff in doing end-of-life care planning with people with intellectual disabilities. Following a trialling process with support staff, the final toolkit was made freely available online. The research team included four researchers with intellectual disabilities (A.C., D.J., L.J., and R.K.-B). Researchers with intellectual disability have been part of every step of the research process; from study design to data collection and analysis to dissemination of study findings.Intellectual disability service provider representatives (M.W., N.P., and S.S.) were part of the co-design group as well. Two of these representatives were also co-applicants in the overall project (N.P. and S.S.). The co-design group included people with intellectual disabilities, families, intellectual disability support staffand health and social care professionals. The study was supported by a Research Advisory Group comprising a variety of stakeholders, including people with intellectual disabilities families, intellectual disability researchers, representatives from intellectual disability organisations,and policymakers.

Reflections on How What We Say, Do, and Acknowledge as Intellectual and Developmental Disability Researchers Matters.

This article highlights reflections by the author on the importance of considering what we say, do and acknowledge in intellectual and developmental disability research. The goal is to advance thinking that can lead to personal and collective change in our approaches to truly share power and elevate the expertise of people with lived experience with intellectual and developmental disabilities in the movement for equity, inclusion, and disability justice. Implications for inclusive research, policy, and practice are discussed as is the need to engage in personal reflection and build new partnerships for collective change.

Why do we continue to exclude the most vulnerable in our society in diabetes research and education? Addressing the challenges presented by people with intellectual disability

Abstract People with an intellectual disability are more likely to develop diabetes compared to their non‐disabled peers. There has been a history of exclusion of people with an intellectual disability from diabetes research and education: we must now challenge this from a human rights perspective. In challenging these perceptions, we must identify the ethical and methodological reasons for exclusion and offer practical solutions to these challenges.These barriers to inclusion focus on three core areas. Firstly, individual factors concerning the person with the disability (can the person give their own informed consent, can the person read/write, completion of self‐report measures?). Secondly, factors concerning the methodology and design of the research study (can studies recruit the numbers needed, can people with intellectual disability understand/accept randomisation?). And, thirdly, system/organisational factors pertaining to the enablers/barriers to engaging in and completing research studies (buy‐in from senior managers, staff acting as gatekeepers, maternalistic attitudes held by gatekeepers, fidelity of intervention delivery).This Janet Kinson paper will explore these intentional and unintentional exclusion criteria that are so often applied to people with an intellectual disability in research trial studies. This paper will dispel the myths and offer solutions for including people with an intellectual disability in diabetes research and education. Using the UK national diabetes structured education programme, DESMOND, this paper will highlight the types of reasonable adjustments that can be made to adapt this programme suitable for adults with an intellectual disability: called DESMOND‐ID. This paper will demonstrate how, with reasonable adjustments, this newly‐adapted DESMOND‐ID education programme then can be tested within a National Institute for Health and Care Research clinical randomised control trial across the UK investigating the programme's clinical and cost‐effectiveness. Copyright © 2024 John Wiley & Sons.

Measurement Invariance in Intellectual and Developmental Disability Research.

Measurement invariance (MI) is a psychometric property of an instrument indicating the degree to which scores from an instrument are comparable across groups. In recent years, there has been a marked uptick in publications using MI in intellectual and developmental disability (IDD) samples. Our goal here is to provide an overview of why MI is important to IDD researchers and to describe some challenges to evaluating it, with an eye towards nudging our subfield into a more thoughtful and measured interpretation of studies using MI.

Clinical and molecular outcomes from the 5-Year natural history study of SSADH Deficiency, a model metabolic neurodevelopmental disorder

BackgroundSuccinic semialdehyde dehydrogenase deficiency (SSADHD) represents a model neurometabolic disease at the fulcrum of translational research within the Boston Children’s Hospital Intellectual and Developmental Disabilities Research Centers (IDDRC), including the NIH-sponsored natural history study of clinical, neurophysiological, neuroimaging, and molecular markers, patient-derived induced pluripotent stem cells (iPSC) characterization, and development of a murine model for tightly regulated, cell-specific gene therapy.MethodsSSADHD subjects underwent clinical evaluations, neuropsychological assessments, biochemical quantification of γ-aminobutyrate (GABA) and related metabolites, electroencephalography (standard and high density), magnetoencephalography, transcranial magnetic stimulation, magnetic resonance imaging and spectroscopy, and genetic tests. This was parallel to laboratory molecular investigations of in vitro GABAergic neurons derived from induced human pluripotent stem cells (hiPSCs) of SSADHD subjects and biochemical analyses performed on a versatile murine model that uses an inducible and reversible rescue strategy allowing on-demand and cell-specific gene therapy.ResultsThe 62 SSADHD subjects [53% females, median (IQR) age of 9.6 (5.4–14.5) years] included in the study had a reported symptom onset at ∼ 6 months and were diagnosed at a median age of 4 years. Language developmental delays were more prominent than motor. Autism, epilepsy, movement disorders, sleep disturbances, and various psychiatric behaviors constituted the core of the disorder’s clinical phenotype. Lower clinical severity scores, indicating worst severity, coincided with older age (R= -0.302, p = 0.03), as well as age-adjusted lower values of plasma γ-aminobutyrate (GABA) (R = 0.337, p = 0.02) and γ-hydroxybutyrate (GHB) (R = 0.360, p = 0.05). While epilepsy and psychiatric behaviors increase in severity with age, communication abilities and motor function tend to improve. iPSCs, which were differentiated into GABAergic neurons, represent the first in vitro neuronal model of SSADHD and express the neuronal marker microtubule-associated protein 2 (MAP2), as well as GABA. GABA-metabolism in induced GABAergic neurons could be reversed using CRISPR correction of the pathogenic variants or mRNA transfection and SSADHD iPSCs were associated with excessive glutamatergic activity and related synaptic excitation.ConclusionsFindings from the SSADHD Natural History Study converge with iPSC and animal model work focused on a common disorder within our IDDRC, deepening our knowledge of the pathophysiology and longitudinal clinical course of a complex neurodevelopmental disorder. This further enables the identification of biomarkers and changes throughout development that will be essential for upcoming targeted trials of enzyme replacement and gene therapy.

The Brain Gene Registry: a data snapshot

Monogenic disorders account for a large proportion of population-attributable risk for neurodevelopmental disabilities. However, the data necessary to infer a causal relationship between a given genetic variant and a particular neurodevelopmental disorder is often lacking. Recognizing this scientific roadblock, 13 Intellectual and Developmental Disabilities Research Centers (IDDRCs) formed a consortium to create the Brain Gene Registry (BGR), a repository pairing clinical genetic data with phenotypic data from participants with variants in putative brain genes. Phenotypic profiles are assembled from the electronic health record (EHR) and a battery of remotely administered standardized assessments collectively referred to as the Rapid Neurobehavioral Assessment Protocol (RNAP), which include cognitive, neurologic, and neuropsychiatric assessments, as well as assessments for attention deficit hyperactivity disorder (ADHD) and autism spectrum disorder (ASD). Co-enrollment of BGR participants in the Clinical Genome Resource’s (ClinGen’s) GenomeConnect enables display of variant information in ClinVar. The BGR currently contains data on 479 participants who are 55% male, 6% Asian, 6% Black or African American, 76% white, and 12% Hispanic/Latine. Over 200 genes are represented in the BGR, with 12 or more participants harboring variants in each of these genes: CACNA1A, DNMT3A, SLC6A1, SETD5, and MYT1L. More than 30% of variants are de novo and 43% are classified as variants of uncertain significance (VUSs). Mean standard scores on cognitive or developmental screens are below average for the BGR cohort. EHR data reveal developmental delay as the earliest and most common diagnosis in this sample, followed by speech and language disorders, ASD, and ADHD. BGR data has already been used to accelerate gene-disease validity curation of 36 genes evaluated by ClinGen’s BGR Intellectual Disability (ID)-Autism (ASD) Gene Curation Expert Panel. In summary, the BGR is a resource for use by stakeholders interested in advancing translational research for brain genes and continues to recruit participants with clinically reported variants to establish a rich and well-characterized national resource to promote research on neurodevelopmental disorders.

Using Inclusive Research Methods and the Housing Pathways Framework in Future Planning and Housing Research: A Pilot Study

Housing and future planning have been key areas of interest in intellectual and development disabilities research for a number of decades. However, the voices of adults with intellectual disabilities are underrepresented in this area of research. Furthermore, the use of inclusive research methods remains limited in the literature. This study sought to pilot the use of inclusive research approaches to investigate the viability of these methods and to begin to build an evidence base of inclusive research in this area of work. Inclusive data analysis and co-authorship approaches were used on a small qualitative dataset from a larger study investigating future planning and transitions out of the family home by adults with intellectual disabilities and their families in Australia. Three semi-structured interviews with adults with intellectual disabilities and family members regarding their housing preferences and planning were analysed using an inclusive data analysis approach following the principles of Interpretative Phenomenological Analysis. These were then further analysed using a plain language version of the housing pathways framework. The results of the pilot study will be used to inform the inclusive research methods used for the remainder of the project dataset. Overall, the use of inclusive methods to pilot a conceptual model to better understand qualitative data was found to be feasible. Small adjustments to the process and accessibility to better support engagement with the research process are recommended. Lastly, greater investigation into co-authorship approaches and options is suggested as a fruitful avenue of inquiry for future research.

Inclusive Dissemination: Inclusive Research Dissemination With Individuals With Intellectual and Developmental Disabilities

Abstract Inclusive research emphasizes the importance of researchers with intellectual and developmental disabilities playing active roles in all phases of research, including research and information dissemination. This article explores how contemporary literature and practical application of inclusive practices in the academic setting of a University Center of Excellence in Developmental Disabilities sheds light on this important yet often overlooked facet of research. It argues that, by adopting a suite of new and more inclusive approaches to dissemination, intellectual and developmental disability research and analysis can reach far beyond the walls of the academy. Approaches to inclusive and strategic sharing of research and other information are presented that can enhance the application and influence of that research. Strategies for increasing inclusivity are also detailed, along with challenges and future considerations to help the field enshrine these strategies in common practice. Finally, barriers to inclusive dissemination are discussed, along with methods to overcome them. Although it is clear that more research and work need to be done on this subject, these strategies, approaches, and methods will help research teams become more inclusive, ensuring the research is meaningful and accessible to people whose lives are affected by it.

Photographic visualization of stories: documenting the experiences of people with intellectual disabilities with guided photovoice.

People with intellectual disabilities are often excluded from participation in research, whilst research has the potential to enhance positive change in their lives. In the current study, using a guided photovoice procedure, the experiences of people with intellectual disabilities regarding participation in research are evaluated. A total of 14 participants with intellectual disabilities were interviewed using guided photovoice. Through thematical analyses the experiences of people with intellectual disabilities regarding photovoice are discussed. Benefits were taking time, visual cues and the opportunity to speak out. Challenges were abstract concepts and aftercare. Guided photovoice helps to better understand the perspective of people with intellectual disabilities and thus can help to improve their quality of life. People with intellectual disabilities were interested in taking part in the current research project and experienced participating as a positive experience. However careful planning and expertise in communication with people with intellectual disabilities is necessary to include people with intellectual disabilities in research.

Photovoice Reimagined: A Guide to Supporting the Participation of Students With Intellectual Disabilities in Research

Photovoice Reimagined: A Guide to Supporting the Participation of Students With Intellectual Disabilities in Research

Advancing Strength-Based Inclusive Mental Health Research in Intellectual and Developmental Disabilities

Advancing Strength-Based Inclusive Mental Health Research in Intellectual and Developmental Disabilities

The role of people with intellectual disability in intellectual disability research: A systematic review of Delphi studies.

The way intellectual disability research is designed warrants critical examination, as the knowledge produced through these approaches informs evidence-based practices. People with lived experience should be considered experts in relation to understanding their bodies, conditions, and treatment. This systematic review analyses the design of Delphi studies, to determine the extent to which people with intellectual disability are involved as experts. The design of Delphi studies (involving structured feedback from experts) provides an insight into the extent that 'lived experience' is valued as a source of expert knowledge. Fifty-five publications reporting on forty-nine separate Delphi studies met our inclusion criteria. Nine publications report the involvement of people with intellectual disability. However, family/informal caregivers are represented as experts in higher numbers and their voices carry greater weight. The findings of this review include guidance for practitioners and researchers to facilitate greater participatory roles of people with intellectual disability.

Reasons for collaborating in inclusive research projects: The perspectives of researchers with experiential knowledge, academic researchers and principal investigators

AbstractInclusive research—in which people with an intellectual disability both collaborate with researchers and work as researchers themselves—has gained increased attention over the last three decades. Although the foundational principles of conducting inclusive research are well‐established at this point, there is a relative dearth of insights concerning the underlying reasons for collaborating as part of inclusive research projects. Therefore, this study sought to identify the reasons why researchers with experiential knowledge (n = 9), academic researchers (n = 8) and principal investigators (n = 10) collaborate within inclusive research projects. All 27 participants were interviewed individually, in order to explore their reasons for collaborating within one of the six inclusive research projects, which encompassed a range of research avenues within the field of intellectual disability research. A thematic analysis was conducted to gain insight into these reasons. For the three groups of participants, several themes emerged, such as experiencing full participation (researchers with experiential knowledge), making research (processes) better suited to the needs of participants with an intellectual disability (academic researchers) and striving for equivalence (principal investigators). Understanding why people collaborate within inclusive research projects is important for facilitating collaborative partnerships, which are a precondition for inclusive research projects.

A Call-In for Allyship and Anti-Ableism in Intellectual Disability Research.

Provoked by decades of grassroots activism, anti-ableist work is underway to advance disability rights. Intellectual disability (ID) researchers can integrate these social changes into their work by interrogating and transforming the beliefs and practices that underpin ID research. We share actionable ideas to foster anti-ableism and allyship in ID research. These include: (1) Learn from and nurture long-term, mutual relationships with people with ID; (2) Amplify the voices of people with ID in institutional structures that influence research; (3) Infuse anti-ableist frameworks into our own research; and (4) Embody a career-long commitment to disability rights, reflexive practice, and growth.

Developing a Patient and Public Involvement Training Course for People with Intellectual Disabilities: The Leicestershire Experience

There is a lack of meaningful involvement of people with intellectual disability in research, outside of the context of study participation. There is a need to develop adapted means of facilitating Patient and Public Involvement when working with people with intellectual disability, that takes account for their level of neurodevelopmental functioning. In this article we describe a Patient and Public Involvement course developed for people with intellectual disability, developed by professionals working with this group, which was subsequently piloted in Leicestershire UK. Different versions of the course were developed for student participants with different levels of neurodevelopmental functioning and communication impairment. For Part 1 of the course (‘What is Research?’), students from Pathway 1 (mild intellectual disability) and Pathway 2 (moderate intellectual disability) both had significantly increased (p ≤ 0.05) mean knowledge test scores post-course relative to pre-course, with non-significant increases in mean confidence test scores for both pathway groups. For Part 2 of the course (‘Your Involvement in Research’), most participants agreed or strongly agreed with the statement ‘I found this interesting’ (Pathway 1: 8 of 9 students [89%]; Pathway 2: 8 of 10 students [80%]), but less students provided equivalent responses to the statement ‘I want to be involved in research in the future’ (Pathway 1: 2 of 9 students [22%]; Pathway 2: 8 of 10 students [80%]). This training course provides a template to provide basic training for people with intellectual disability with respect to research involvement, but requires further evaluation in larger student samples of diverse demographic and neurodevelopmental characteristics.

The Work Ahead for Intellectual and Developmental Disabilities Research.

In "Toward Equity in Research on Intellectual and Developmental Disabilities," we sought to make entrenched assumptions and practices of intellectual and developmental disabilities research visible by explicitly describing the status quo in terms of models of disability, participant and researcher identities, research priorities, and biases in measurement and treatment approaches. We then curated individual- and systems-level actions drawn from disability justice and broader social justice lenses to offer a way forward. We focused on three major areas (i.e., intersectionality and person-centered approaches, participatory research, and interprofessional collaboration), depicting influences, methods, and actions in a framework of disability, identity, and culture. In this Author Response, we address five commentaries that critique and extend that synthesis.

The Equity Agenda in Intellectual and Developmental Disabilities Research.

This commentary on Kover and Abbeduto (2023) underscores the critical importance of naming and framing toward an equity agenda in intellectual and developmental disabilities research. More specifically, I briefly outline (1) why racialization is an important anchor in IDD discourse; (2) whiteness as a necessary point of discussion; and (3) the adoption of critical inquiry and critical praxis.

Resilience in the face of adversity: How people with intellectual disabilities deal with challenging times.

In people with intellectual disabilities research and policy are often still focused on risks or the prevention of risks. Research on the process of resilience is in its infancy in the care for people with intellectual disabilities. In the current study, applying a guided photovoice procedure, people with intellectual disabilities were asked what helped them to deal with adverse events. Additionally, informants from their social network were asked to reflect on this question. The following sources of resilience were identified: acceptance, autonomy, beautiful memories, perseverance, physical wellbeing, positive emotions, social skills, spirituality, activities, a home and the social network. Our findings provide practical guidelines for clinicians to talk about resilience with people with intellectual disabilities. Suggestions for future research are made that will contribute to the process of resilience and inclusion of people with intellectual disabilities.

Behavioural sleep problems in children and adults with intellectual disabilities: An integrative literature review.

People with intellectual disabilities are more likely to experience sleep problems, which can affect quality of life, physical health, mental health and well-being. An integrative literature review was conducted to investigate what is known about behavioural sleep disturbances in people with an intellectual disability. The search used the following databases: Scopus, PsycInfo and Cinahl, to find papers published since 2015. Within intellectual disability research, sleep appears as a common issue due to its high prevalence, negative relationships with an individual's physical and mental health, their quality of life, and impact of sleep problems on family or carers. The growing evidence base appears to support the use of behavioural, lifestyle and pharmacological interventions to improve sleep in people with an intellectual disability. A wide array of literature provides evidence that people with intellectual disabilities are affected by and need support with their sleep.