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Tuesday, March 24, 2020
Researchers develop language test for people with Fragile X syndrome
NIH-funded test seeks to increase participation of individuals with Fragile X in clinical trials.
Researchers have developed a test to measure the expressive language skills of people with Fragile X syndrome, a genetic disorder that may result in intellectual disability, cognitive impairment and symptoms of autism spectrum disorder. Expressive language refers to the use of words to convey meaning to others. The work was funded by the Eunice Kennedy Shriver National Institute of Child Health and Human Development, part of the National Institutes of Health.
The researchers developed the test to provide an objective way to measure improvement in language, which may help increase the participation of people with Fragile X in clinical studies aimed at improving intellectual and cognitive functioning. The researchers were led by Leonard Abbeduto, Ph.D., of the University of California, Davis MIND Institute. It appears in the Journal of Neurodevelopmental Disorders.
The test consists of two parts. In the first, participants engage in a conversation with the test administrator on topics that interest them, including their favorite activities, famous people or fictional characters, familiar people, and specific events or themes. In the second part, participants narrate the events in a wordless picture book. Their responses are recorded and the transcripts are scored on such aspects of expressive language as talkativeness, how they arrange words to form sentences, whether they use parts of speech correctly, and how understandable their speech is.
Researchers administered the test to 106 individuals with Fragile X syndrome and intellectual disability, from ages 6 to 23. Participants took the test once and again four weeks later. Test score were consistent across both versions, which showed that the test was reliable. Except for participants under 12, test scores were consistent with expressive language scores on other tests of cognitive skills the participants took earlier, indicating the reliability of the test measurements. Similarly, noncompliance—not cooperating with the test administrator—was also highest for this age group. The researchers concluded that test measures were appropriate for people with Fragile X syndrome over age 12.
Funding was provided by NIH’s Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) and National Center for Advancing Translational Sciences.
Alice Kau, Ph.D., NICHD Intellectual and Developmental Disabilities Branch, project officer for the study, is available for comment.
Abbeduto, L. Expressive language sampling as a source of outcome measures for treatment studies in fragile X syndrome: feasibility, practice effects, test-retest reliability, and construct validity. Journal of Neurodevelopmental Disorders. 2020. https://doi.org/10.1186/s11689-020-09313-6.
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