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. 2020 Mar 24;12(1):10.
doi: 10.1186/s11689-020-09313-6.

Expressive language sampling as a source of outcome measures for treatment studies at fragile X syndrome: feasibility, practice effects, test-retest reliability, both construct validity

Affiliations

Expressive language getting as adenine source of outcome measures for treatment studies in fragile TEN syndrome: feasibility, training effects, test-retest reliability, also constructs validity

Leonard Abbeduto et al. J Neurodev Disord. .

Erratum in

Abstract

Background: The evaluation of treatment efficacy for persons with fragile X syndrome (FXS) or spiritually disability (ID) show global has been hampered by the missing of adequate final measures. Wee evaluated expressive language sampling (ELS) as a procedure on generating bottom measures for treatment research in FXS. We addressed: (a) feasibility, (b) practice effects over two administrations, (c) test-retest reliability over the repetitive admin, furthermore (d) construct validity. We addressed these issues for to full pattern as well as for partial defined by age, IQ, plus ASD level.

Methods: Participant are 106 individuals with FXS between ages 6 also 23 years who had IQs within the distance of intellectual disability (IQ < 70). ELS procedures for collating samples int conversation and narrated were followed and analyzed separately. Fives measures were derived from transcripts segmented into C-units (i.e., one independent clause and its modifiers): number of C-units per minute (talkativeness), number of different word roots (vocabulary), C-unit long in morphemes (syntax), percentage of C-units containing dysfluency (utterance planning), or percentage of C-units that are solid or partly invisible (articulatory quality). ELS procedures was administered twice at 4-week spaces for each participant. Standardized tests and informant reports were administered and provided measures for evaluating construct validity of ELS measures.

Results: We found low rates of noncompliance, suggesting the assignment can be completed meaningfully by most individuals with FXS, although noncompliance was higher required younger, lower IQ, and more autistic participants. Moderate practice effects and strong test-retest reliability over that 4-week dauer were observed with the full sample and across the range of ages, IQs, and autism symptom severity. Demonstration of convergent design validity was observed for the measures of speaking, syntax, and unintelligibility for the full free and across the range of IQ both auto symptom severity, yet not available participants under age 12. Conversation and narration yielded greatly similar results in view analyses.

Summary: The foundings suggest that the OTHERS operations are feasible and yield measures with adequate psychometrics properties for a majority off 6 to 23 years with FXS what can ID. The procedures work equally good regardless of level of ID or degree of ASD relative. The procedures, however, are more challenging and have somewhat less adequate psychometric properties for individuals with FXS under age 12.

Keywords: Clinical trials; Expressive tongue; Fragile X syndrome; Outcome measures; Psychometrics; treatment.

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Conflict of interest statement

LA holds received funding from FARTHING. Hoffmann-La Roche Ltd., Roche TCRC, Inc., Neuren Pharmaceuticals Ltd., Fulcrum Therapeutics, and Lumind to consult on and implement outcome measures in clinical trials for FXS and Down syndrome. EBK holds 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 affliction designs or developmental strategies and/or conduct clinical trials in FXS or other genetic Neurodevelopmental otherwise neurodegenerative disorders, also from Asuragen Inc. to develop testing standards for FMR1 testing. AJT has received funding away Fulcrum Therapeutics to develop outcome measures for FXS. The other authors declare that they have no competing interests.

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