Behavioural Research in Intellectual disability Development and Growth Evaluation- Measuring cognitive outcomes in people with neurogenetic syndromes associated with an intellectual disability
Year: 2026
Type: preprint
Abstract: Intellectual disabilities (ID) affect approximately 1% of the population, with up to half of ID diagnoses attributable to neurogenetic syndromes, such as Down syndrome, fragile X syndrome, and Angelman syndrome. Evidence suggests that people with neurogenetic syndromes associated with ID often experience executive function differences, such as difficulties with working memory, inhibition, and cognitive flexibility, which can influence learning, behaviour, wellbeing, and independence. Despite this, people with neurogenetic syndromes associated with ID are frequently excluded from cognitive and clinical research. A key barrier is the lack of developmentally appropriate, psychometrically robust, and accessible assessment tools. Existing measures are often not validated for people with ID, particularly those with more severe-to-profound ID, and may fail to capture the full range of ability. In-person assessments can also introduce barriers, including fatigue, anxiety, and accessibility challenges. Remote, play-based approaches offer a promising alternative by reducing burden and enabling assessments in familiar environments, yet their feasibility and validity remain underexplored. Understanding feasibility will also greatly improve the ability to measure and assess executive function skills in a population that is often excluded due to co-occurring ID. This study therefore evaluates a novel remote, play-based battery of executive function tasks for people with neurogenetic syndromes associated with ID, examining feasibility, acceptability, test–retest reliability, and convergent validity with a pre-existing battery, and comparing parent-led and researcher-led administration. Improved understanding and measurement of executive function in these populations can inform assessment, intervention, and support strategies, ultimately prompting more tailored and inclusive approaches to education and interventions.
Language: English
Field: Medicine
Domain: Health Sciences
Open Access status: green
OpenAlex ID: W7148580303