The NIH Toolbox Cognitive Battery for intellectual disabilities: three preliminary studies and future directions
| Autor: | Elizabeth Berry-Kravis, Keith F. Widaman, Jeanine Coleman, Stephanie M. Sansone, Leonard J Abbeduto, Andrea Schneider, Richard Gershon, Kelly C. Rhodes, David R Hessl, Karen Riley, Dena Oaklander |
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| Rok vydání: | 2016 |
| Předmět: |
Ecological validity
Intellectual and Developmental Disabilities (IDD) Cognitive Neuroscience Down syndrome Context (language use) NIH Toolbox Assessment Outcome measures Basic Behavioral and Social Science Pathology and Forensic Medicine 03 medical and health sciences 0302 clinical medicine Cognition FMR1 gene Rare Diseases New Method Clinical Research Behavioral and Social Science Psychology 0501 psychology and cognitive sciences Intellectual and Developmental Disabilities Mental age Pediatric 05 social sciences Neuropsychology Neurosciences Construct validity 3. Good health Brain Disorders Good Health and Well Being Mental Health Convergent validity Fragile X Syndrome Neurological Pediatrics Perinatology and Child Health Neurology (clinical) 030217 neurology & neurosurgery 050104 developmental & child psychology Clinical psychology Fragile X syndrome |
| Zdroj: | Journal of neurodevelopmental disorders, vol 8, iss 1 Journal of Neurodevelopmental Disorders |
| Popis: | BackgroundRecent advances in understanding molecular and synaptic mechanisms of intellectual disabilities (ID) in fragile X syndrome (FXS) and Down syndrome (DS) through animal models have led to targeted controlled trials with pharmacological agents designed to normalize these underlying mechanisms and improve clinical outcomes. However, several human clinical trials have failed to demonstrate efficacy of these targeted treatments to improve surrogate behavioral endpoints. Because the ultimate index of disease modification in these disorders is amelioration of ID, the validation of cognitive measures for tracking treatment response is essential. Here, we present preliminary research to validate the National Institutes of Health Toolbox Cognitive Battery (NIH-TCB) for ID.MethodsWe completed three pilot studies of patients with FXS (total n = 63; mean age 19.3 ± 8.3years, mean mental age 5.3 ± 1.6years), DS (n = 47; mean age 16.1 ± 6.2, mean mental age 5.4 ± 2.0), and idiopathic ID (IID; n = 16; mean age 16.1 ± 5.0, mean mental age 6.6 ± 2.3) measuring processing speed, executive function, episodic memory, word/letter reading, receptive vocabulary, and working memory using the web-based NIH-TB-CB, addressing feasibility, test-retest reliability, construct validity, ecological validity, and syndrome differences and profiles.ResultsFeasibility was good to excellent (≥80% of participants with valid scores) for above mental age 4years for all tests except list sorting (working memory). Test-retest stability was good to excellent, and convergent validity was similar to or better than results obtained from typically developing children in the normal sample for executive function and language measures. Examination of ecological validity revealed moderate to very strong correlations between the NIH-TCB composite and adaptive behavior and full-scale IQ measures. Syndrome/group comparisons demonstrated significant deficits for the FXS and DS groups relative to IID on attention and inhibitory control, a significant reading weakness for FXS, and a receptive vocabulary weakness for DS.ConclusionsThe NIH-TCB has potential for assessing important dimensions of cognition in persons with ID, and several tests may be useful for tracking response to intervention. However, more extensive psychometric studies, evaluation of the NIH-TCB's sensitivity to change, both developmentally and in the context of treatment, and perhaps establishing links to brain function in these populations, are required to determine the true utility of the battery as a set of outcome measures. |
| Databáze: | OpenAIRE |
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