Dataset Information

Signaling noncomprehension of language: a comparison of fragile X syndrome and Down syndrome.

ABSTRACT: Signaling noncomprehension of the spoken messages of others was examined for youth with fragile X or Down syndrome in comparison with each other and nonverbal MA-matched typically developing children. A direction-following task was used in which some of the directions were inadequate. Both syndrome groups signaled noncomprehension less often than did the typically developing children. The ability to signal noncomprehension appropriately was related to a measure of receptive vocabulary and syntax. Preliminary analyses indicated that males with fragile X syndrome signaled noncomprehension less often than did their female peers, even after controlling for differences in nonverbal MA.

SUBMITTER: Abbeduto L

PROVIDER: S-EPMC5610960 | biostudies-literature | 2008 May

REPOSITORIES: biostudies-literature

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Signaling noncomprehension of language: a comparison of fragile X syndrome and Down syndrome.

Abbeduto Leonard L Murphy Melissa M MM Kover Sara T ST Giles Nancy D ND Karadottir Selma S Amman Adrienne A Bruno Loredana L Kim Jee-Seon JS Schroeder Susen S Anderson Julie A JA Nollin Kathryn A KA

American journal of mental retardation : AJMR 20080501 3

Signaling noncomprehension of the spoken messages of others was examined for youth with fragile X or Down syndrome in comparison with each other and nonverbal MA-matched typically developing children. A direction-following task was used in which some of the directions were inadequate. Both syndrome groups signaled noncomprehension less often than did the typically developing children. The ability to signal noncomprehension appropriately was related to a measure of receptive vocabulary and syntax ...[more]

PMID: 18407723

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Project description:BACKGROUNDFXLEARN, the first-ever large multisite trial of effects of disease-targeted pharmacotherapy on learning, was designed to explore a paradigm for measuring effects of mechanism-targeted treatment in fragile X syndrome (FXS). In FXLEARN, the effects of metabotropic glutamate receptor type 5 (mGluR5) negative allosteric modulator (NAM) AFQ056 on language learning were evaluated in 3- to 6-year-old children with FXS, expected to have more learning plasticity than adults, for whom prior trials of mGluR5 NAMs have failed.METHODSAfter a 4-month single-blind placebo lead-in, participants were randomized 1:1 to AFQ056 or placebo, with 2 months of dose optimization to the maximum tolerated dose, then 6 months of treatment during which a language-learning intervention was implemented for both groups. The primary outcome was a centrally scored videotaped communication measure, the Weighted Communication Scale (WCS). Secondary outcomes were objective performance-based and parent-reported cognitive and language measures.RESULTSFXLEARN enrolled 110 participants, randomized 99, and had 91 who completed the placebo-controlled period. Although both groups made language progress and there were no safety issues, the change in WCS score during the placebo-controlled period was not significantly different between the AFQ056 and placebo-treated groups, nor were there any significant between-group differences in change in any secondary measures.CONCLUSIONDespite the large body of evidence supporting use of mGluR5 NAMs in animal models of FXS, this study suggests that this mechanism of action does not translate into benefit for the human FXS population and that better strategies are needed to determine which mechanisms will translate from preclinical models to humans in genetic neurodevelopmental disorders.TRIAL REGISTRATIONClincalTrials.gov NCT02920892.FUNDING SOURCESNeuroNEXT network NIH grants U01NS096767, U24NS107200, U24NS107209, U01NS077323, U24NS107183, U24NS107168, U24NS107128, U24NS107199, U24NS107198, U24NS107166, U10NS077368, U01NS077366, U24NS107205, U01NS077179, and U01NS077352; NIH grant P50HD103526; and Novartis IIT grant AFQ056X2201T for provision of AFQ056.

Dataset Information

Signaling noncomprehension of language: a comparison of fragile X syndrome and Down syndrome.

Publications

Signaling noncomprehension of language: a comparison of fragile X syndrome and Down syndrome.

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