Author:
Abbeduto Leonard,del Hoyo Soriano Laura,Berry-Kravis Elizabeth,Sterling Audra,Edgin Jamie O.,Abdelnur Nadia,Drayton Andrea,Hoffmann Anne,Hamilton Debra,Harvey Danielle J.,Thurman Angela John
Abstract
AbstractThe lack of psychometrically sound outcome measures has been a barrier to evaluating the efficacy of treatments proposed for core symptoms of intellectual disability (ID). Research on Expressive Language Sampling (ELS) procedures suggest it is a promising approach to measuring treatment efficacy. ELS entails collecting samples of a participant’s talk in interactions with an examiner that are naturalistic but sufficiently structured to ensure consistency and limit examiner effects on the language produced. In this study, we extended previous research on ELS by analyzing an existing dataset to determine whether psychometrically adequate composite scores reflecting multiple dimensions of language can be derived from ELS procedures administered to 6- to 23-year-olds with fragile X syndrome (n = 80) or Down syndrome (n = 78). Data came from ELS conversation and narration procedures administered twice in a 4-week test–retest interval. We found that several composites emerged from variables indexing syntax, vocabulary, planning processes, speech articulation, and talkativeness, although there were some differences in the composites for the two syndromes. Evidence of strong test–retest reliability and construct validity of two of three composites were obtained for each syndrome. Situations in which the composite scores would be useful in evaluating treatment efficacy are outlined.
Funder
National Institutes of Health
Publisher
Springer Science and Business Media LLC
Reference67 articles.
1. Van Naarden Braun, K. et al. Trends in the prevalence of autism spectrum disorder, cerebral palsy, hearing loss, intellectual disability, and vision impairment, Metropolitan Atlanta, 1991–2010. PLoS ONE 10(4), e0124120. https://doi.org/10.1371/journal.pone.0124120 (2015).
2. Dykens, E. M. Behavioral phenotypes and genomic testing: Scientific and societal implications in Handbook of Intellectual and Developmental Disabilities, L. M. Glidden, L. Abbeduto, L. McIntyre, and M. Tasee, Eds. Washington, DC: American Psychological Association Press.
3. Berry-Kravis, E. M. et al. Drug development for neurodevelopmental disorders: Lessons learned from fragile X syndrome. Nat. Rev. Drug Discov. 17(4), 280–299. https://doi.org/10.1038/nrd.2017.221 (2018).
4. Esbensen, A. J. et al. Outcome measures for clinical trials in down syndrome. Am. J. Intellect. Dev. Disabil. 122, 3. https://doi.org/10.1352/1944-7558-122.3.247 (2017).
5. Hagerman, R. & Tuchman, R. Turning the tide on targeted treatments for neurodevelopmental disorders. Neurology 92(16), 741–742. https://doi.org/10.1212/WNL.0000000000007301 (2019).
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