[article]
| Titre : |
Spoken language outcome measures for treatment studies in Down syndrome: feasibility, practice effects, test-retest reliability, and construct validity of variables generated from expressive language sampling |
| Type de document : |
texte imprimé |
| Auteurs : |
Angela John THURMAN, Auteur ; Jamie O. EDGIN, Auteur ; Stephanie L SHERMAN, Auteur ; Audra STERLING, Auteur ; Andrea MCDUFFIE, Auteur ; Elizabeth BERRY-KRAVIS, Auteur ; Debra HAMILTON, Auteur ; Leonard ABBEDUTO, Auteur |
| Langues : |
Anglais (eng) |
| Mots-clés : |
Adolescent Adult Child Child, Preschool Down Syndrome Feasibility Studies Humans Language Outcome Assessment, Health Care Reproducibility of Results Young Adult Clinical trials Down syndrome Expressive language Expressive language sampling Outcome measures Psychometrics Treatment Pharmaceuticals Ltd., Fulcrum Therapeutics, and Lumind to consult on and implement outcome measures in clinical trials for FXS and DS. EBK has received funding from Seaside Therapeutics, Novartis, Roche, Alcobra, Neuren, Cydan, Fulcrum, GW, Neurotrope, Marinus, Zynerba, BioMarin, Lumos, Ovid, AMO, Yamo, Ionis, GeneTx, Acadia, Neurogene, Ultragenyx, and Vtesse/Sucampo/Mallinkcrodt Pharmaceuticals to consult on trial design or development strategies and/or conduct clinical trials in FXS or other genetic neurodevelopmental or neurodegenerative disorders, and from Asuragen Inc. to develop testing standards for FMR1 testing. AJT has received funding from Fulcrum Therapeutics to develop outcome measures for FXS. The other authors declare that they have no competing interests. |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: The purpose of this study was to evaluate expressive language sampling (ELS) as a procedure for generating spoken language outcome measures for treatment research in Down syndrome (DS). We addressed (a) feasibility, (b) practice effects across two short-term administrations, (c) test-retest reliability across two short-term administrations, (d) convergent and discriminant construct validity, and (e) considered comparisons across the conversation and narration contexts. METHOD: Participants were 107 individuals with DS between 6 and 23 years of age who presented with intellectual disability (IQ < 70). The utility of ELS procedures designed to collect samples of spoken language in conversation and narration were evaluated separately. Variables of talkativeness, vocabulary, syntax, utterance planning, and articulation quality, derived from transcripts segmented into C-units (i.e., an independent clause and its modifiers), were considered. A 4-week interval was used to assess practice effects and test-retest reliability. Standardized direct assessments and informant report measures were collected to evaluate construct validity of the ELS variables. RESULTS: Low rates of noncompliance were observed; youth who were under 12 years of age, had phrase-level speech or less, and had a 4-year-old developmental level or less were at particular risk for experiencing difficulty completing the ELS procedures. Minimal practice effects and strong test-retest reliability across the 4-week test-retest interval was observed. The vocabulary, syntax, and speech intelligibility variables demonstrated strong convergent and discriminant validity. Although significant correlations were found between the variables derived from both the conversation and narration contexts, some differences were noted. CONCLUSION: The ELS procedures considered were feasible and yielded variables with adequate psychometric properties for most individuals with DS between 6 and 23 years old. That said, studies of outcome measures appropriate for individuals with DS with more limited spoken language skills are needed. Context differences were observed in ELS variables suggest that comprehensive evaluation of expressive language is likely best obtained when utilizing both contexts. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-021-09361-6 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 |
in Journal of Neurodevelopmental Disorders > 13 (2021)
[article] Spoken language outcome measures for treatment studies in Down syndrome: feasibility, practice effects, test-retest reliability, and construct validity of variables generated from expressive language sampling [texte imprimé] / Angela John THURMAN, Auteur ; Jamie O. EDGIN, Auteur ; Stephanie L SHERMAN, Auteur ; Audra STERLING, Auteur ; Andrea MCDUFFIE, Auteur ; Elizabeth BERRY-KRAVIS, Auteur ; Debra HAMILTON, Auteur ; Leonard ABBEDUTO, Auteur. Langues : Anglais ( eng) in Journal of Neurodevelopmental Disorders > 13 (2021)
| Mots-clés : |
Adolescent Adult Child Child, Preschool Down Syndrome Feasibility Studies Humans Language Outcome Assessment, Health Care Reproducibility of Results Young Adult Clinical trials Down syndrome Expressive language Expressive language sampling Outcome measures Psychometrics Treatment Pharmaceuticals Ltd., Fulcrum Therapeutics, and Lumind to consult on and implement outcome measures in clinical trials for FXS and DS. EBK has received funding from Seaside Therapeutics, Novartis, Roche, Alcobra, Neuren, Cydan, Fulcrum, GW, Neurotrope, Marinus, Zynerba, BioMarin, Lumos, Ovid, AMO, Yamo, Ionis, GeneTx, Acadia, Neurogene, Ultragenyx, and Vtesse/Sucampo/Mallinkcrodt Pharmaceuticals to consult on trial design or development strategies and/or conduct clinical trials in FXS or other genetic neurodevelopmental or neurodegenerative disorders, and from Asuragen Inc. to develop testing standards for FMR1 testing. AJT has received funding from Fulcrum Therapeutics to develop outcome measures for FXS. The other authors declare that they have no competing interests. |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: The purpose of this study was to evaluate expressive language sampling (ELS) as a procedure for generating spoken language outcome measures for treatment research in Down syndrome (DS). We addressed (a) feasibility, (b) practice effects across two short-term administrations, (c) test-retest reliability across two short-term administrations, (d) convergent and discriminant construct validity, and (e) considered comparisons across the conversation and narration contexts. METHOD: Participants were 107 individuals with DS between 6 and 23 years of age who presented with intellectual disability (IQ < 70). The utility of ELS procedures designed to collect samples of spoken language in conversation and narration were evaluated separately. Variables of talkativeness, vocabulary, syntax, utterance planning, and articulation quality, derived from transcripts segmented into C-units (i.e., an independent clause and its modifiers), were considered. A 4-week interval was used to assess practice effects and test-retest reliability. Standardized direct assessments and informant report measures were collected to evaluate construct validity of the ELS variables. RESULTS: Low rates of noncompliance were observed; youth who were under 12 years of age, had phrase-level speech or less, and had a 4-year-old developmental level or less were at particular risk for experiencing difficulty completing the ELS procedures. Minimal practice effects and strong test-retest reliability across the 4-week test-retest interval was observed. The vocabulary, syntax, and speech intelligibility variables demonstrated strong convergent and discriminant validity. Although significant correlations were found between the variables derived from both the conversation and narration contexts, some differences were noted. CONCLUSION: The ELS procedures considered were feasible and yielded variables with adequate psychometric properties for most individuals with DS between 6 and 23 years old. That said, studies of outcome measures appropriate for individuals with DS with more limited spoken language skills are needed. Context differences were observed in ELS variables suggest that comprehensive evaluation of expressive language is likely best obtained when utilizing both contexts. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-021-09361-6 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 |
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