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Gepubliceerd in: Journal of Autism and Developmental Disorders 9/2008

01-10-2008 | Original Paper

Characterization of Potential Outcome Measures for Future Clinical Trials in Fragile X Syndrome

Auteurs: Elizabeth Berry-Kravis, Allison Sumis, Ok-Kyung Kim, Rebecca Lara, Joanne Wuu

Gepubliceerd in: Journal of Autism and Developmental Disorders | Uitgave 9/2008

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Abstract

Clinical trials targeting recently elucidated synaptic defects in fragile X syndrome (FXS) will require outcome measures capable of assessing short-term changes in cognitive functioning. Potentially useful measures for FXS were evaluated here in a test–retest setting in males and females with FXS (N = 46). Good reproducibility, determined by an interclass correlation (ICC) or weighted kappa (κ) of 0.7–0.9 was seen for RBANS List and Story Memory, NEPSY Tower, Woodcock–Johnson Spatial Relations and the commissions score from the Carolina Fragile X Project Continuous Performance Test (CPT). This study demonstrates the feasibility of generating test profiles containing reliability data, ability levels required for test performance, and refusal rates to assist with choice of outcome measures in FXS and other cohorts with cognitive disability.
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Metagegevens
Titel
Characterization of Potential Outcome Measures for Future Clinical Trials in Fragile X Syndrome
Auteurs
Elizabeth Berry-Kravis
Allison Sumis
Ok-Kyung Kim
Rebecca Lara
Joanne Wuu
Publicatiedatum
01-10-2008
Uitgeverij
Springer US
Gepubliceerd in
Journal of Autism and Developmental Disorders / Uitgave 9/2008
Print ISSN: 0162-3257
Elektronisch ISSN: 1573-3432
DOI
https://doi.org/10.1007/s10803-008-0564-8

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