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Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties

The 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. EL...

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Autores principales: 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
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10247708/
https://www.ncbi.nlm.nih.gov/pubmed/37286643
http://dx.doi.org/10.1038/s41598-023-36087-3
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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
author_facet 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
author_sort Abbeduto, Leonard
collection PubMed
description The 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.
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spelling pubmed-102477082023-06-09 Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties 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 Sci Rep Article The 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. Nature Publishing Group UK 2023-06-07 /pmc/articles/PMC10247708/ /pubmed/37286643 http://dx.doi.org/10.1038/s41598-023-36087-3 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Article
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
Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties
title Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties
title_full Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties
title_fullStr Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties
title_full_unstemmed Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties
title_short Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties
title_sort expressive language sampling and outcome measures for treatment trials in fragile x and down syndromes: composite scores and psychometric properties
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10247708/
https://www.ncbi.nlm.nih.gov/pubmed/37286643
http://dx.doi.org/10.1038/s41598-023-36087-3
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