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Expressive language profiles of verbally expressive adolescents and young adults with Down syndrome or fragile X syndrome.

ABSTRACT: In this study, the authors examined the expressive language abilities of a subset of highly verbally expressive adolescents and young adults with Down syndrome (DS) and those with fragile X syndrome (FXS) for evidence of syndrome-related differences. FXS gender differences were also examined in an exploratory fashion.The authors evaluated 24 adolescents and young adults with DS, 17 adolescents and young adults with FXS, and 21 children with typical development (TD), with the groups matched on nonverbal mental age. Language ability was examined using the Oral and Written Language Scales (OWLS; Carrow-Woolfolk, 1995) and Developmental Sentence Scoring (DSS; Lee, 1974) scores derived from an oral narrative language sample.Study analyses revealed the following group differences: The FXS group outperformed the DS and TD groups on the OWLS measure; the TD group outperformed both other groups on some of the DSS measures; the FXS group outperformed the DS group on the DSS Sentence Point measure; and females with FXS outperformed males with FXS on several measures.Results contribute to the ongoing construction of the language phenotypes of individuals with DS and individuals with FXS and support the conclusion that there are quantitative rather than qualitative differences in their expressive language profiles.

SUBMITTER: Finestack LH

PROVIDER: S-EPMC2948067 | biostudies-other | 2010 Oct

REPOSITORIES: biostudies-other

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Expressive language profiles of verbally expressive adolescents and young adults with Down syndrome or fragile X syndrome.

Finestack Lizbeth H LH Abbeduto Leonard L

Journal of speech, language, and hearing research : JSLHR 20100719 5

<h4>Purpose</h4>In this study, the authors examined the expressive language abilities of a subset of highly verbally expressive adolescents and young adults with Down syndrome (DS) and those with fragile X syndrome (FXS) for evidence of syndrome-related differences. FXS gender differences were also examined in an exploratory fashion.<h4>Method</h4>The authors evaluated 24 adolescents and young adults with DS, 17 adolescents and young adults with FXS, and 21 children with typical development (TD) ...[more]

PMID: 20643789

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Project description:ObjectiveTo assess whether parent training in behavioral intervention, combined with a 16-session nutrition and activity education program, would improve weight loss relative to nutrition and activity education alone in adolescents and young adults with Down syndrome.Study designTwenty-one patients with Down syndrome aged 13-26 years with a body mass index ? 85 th percentile were enrolled and randomized to a 6-month nutrition and activity education intervention (n = 10) or to a nutrition and activity education+behaviorial intervention (n = 11), and followed for 6 months after the active intervention period (1-year follow-up). The primary outcome measure was body weight; secondary outcomes included percentage body fat by bioelectric impedance; intake of fruits, vegetables, and energy-dense low-nutrient snack food (treats) by 3-day food record; and moderate/vigorous physical activity by accelerometry.ResultsAt 6 months, mean body weight in the nutrition and activity education+behavioral intervention group was 3.2 kg lower than that in the nutrition and activity education group (95% CI, 1.0-5.5 kg; P = .005). Mean group differences were sustained at 1 year (3.6 kg; 95% CI, 1.4-5.9 kg; P = .002). At 6 months, moderate/vigorous physical activity time increased by an average of 18 minutes/day compared with baseline in the nutrition and activity education+behavioral intervention group (P = .01) and decreased by 7 minutes/day in the nutrition and activity education group (P = .30). These changes were largely maintained at 1 year, but were not statistically significant. Vegetable intake in the nutrition and activity education+behavioral intervention group exceeded that in the nutrition and activity education group by a mean of 1.6 servings at 1 year (P = .009), but not at 6 months. No between-group differences were observed for percentage body fat or consumption of fruits or treats.ConclusionParent-supported behavioral intervention appears to be a successful adjunct to a 6-month nutrition education intervention in achieving weight loss in adolescents and young adults with Down syndrome.

Project description:BackgroundThe lack of robust and reliable clinical biomarkers in Fragile X Syndrome (FXS), the most common inherited form of intellectual disability, has limited the successful translation of bench-to-bedside therapeutics. While numerous drugs have shown promise in reversing synaptic and behavioral phenotypes in mouse models of FXS, none have demonstrated clinical efficacy in humans. Electroencephalographic (EEG) measures have been identified as candidate biomarkers as EEG recordings of both adults with FXS and mouse models of FXS consistently exhibit alterations in resting state and task-related activity. However, the developmental timing of these EEG differences is not known as thus far EEG studies have not focused on young children with FXS. Further, understanding how EEG differences are associated with core symptoms of FXS is crucial to successful use of EEG as a biomarker, and may improve our understanding of the disorder.MethodsResting-state EEG was collected from FXS boys with full mutation of Fmr1 (2.5-7 years old, n = 11) and compared with both age-matched (n = 12) and cognitive-matched (n = 12) typically developing boys. Power spectra (including aperiodic and periodic components) were compared using non-parametric cluster-based permutation testing. Associations between 30 and 50 Hz gamma power and cognitive, language, and behavioral measures were evaluated using Pearson correlation and linear regression with age as a covariate.ResultsFXS participants showed increased power in the beta/gamma range (~ 25-50 Hz) across multiple brain regions. Both a reduction in the aperiodic (1/f) slope and increase in beta/gamma periodic activity contributed to the significant increase in high-frequency power. Increased gamma power, driven by the aperiodic component, was associated with better language ability in the FXS group. No association was observed between gamma power and parent report measures of behavioral challenges, sensory hypersensitivities, or adaptive behaviors.LimitationsThe study sample size was small, although comparable to other human studies in rare-genetic disorders. Findings are also limited to males in the age range studied.ConclusionsResting-state EEG measures from this study in young boys with FXS identified similar increases in gamma power previously reported in adults and mouse models. The observed positive association between resting state aperiodic gamma power and language development supports hypotheses that alterations in some EEG measures may reflect ongoing compensatory mechanisms.

Dataset Information

Expressive language profiles of verbally expressive adolescents and young adults with Down syndrome or fragile X syndrome.

Publications

Expressive language profiles of verbally expressive adolescents and young adults with Down syndrome or fragile X syndrome.

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