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Comparing Tense and Agreement Productivity in Boys With Fragile X Syndrome, Children With Developmental Language Disorder, and Children With Typical Development.

ABSTRACT: Purpose This study compared and characterized the tense and agreement productivity of boys with fragile X syndrome (FXS), children with developmental language disorder (DLD), and children with typical development (TD) matched on mean length of utterance. Method Twenty-two boys with FXS (M age = 12.22 years), 19 children with DLD (M age = 4.81 years), and 20 children with TD (M age = 3.23 years) produced language samples that were coded for their productive use of five tense markers (i.e., third-person singular, past tense -ed, copula BE, auxiliary BE, and auxiliary DO) using the tense and agreement productivity score. Children also completed norm-referenced cognitive and linguistic assessments. Results Children with DLD generally used tense and agreement markers less productively than children with TD, particularly third-person singular and auxiliary BE. However, boys with FXS demonstrated a more complicated pattern of productivity, where they were similar to children with DLD and TD, depending on the tense marker examined. Results revealed that children with DLD and TD showed a specific developmental sequence of the individual tense markers that aligns with patterns documented by previous studies, whereas boys with FXS demonstrated a more even profile of productivity. Conclusions These findings help to further clarify areas of overlap and discrepancy in tense and agreement productivity among boys with FXS and children with DLD. Additional clinical implications of these results are discussed.

SUBMITTER: Hilvert E

PROVIDER: S-EPMC7242987 | biostudies-literature | 2020 Apr

REPOSITORIES: biostudies-literature

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Comparing Tense and Agreement Productivity in Boys With Fragile X Syndrome, Children With Developmental Language Disorder, and Children With Typical Development.

Hilvert Elizabeth E Hoover Jill J Sterling Audra A Schroeder Susen S

Journal of speech, language, and hearing research : JSLHR 20200413 4

Purpose This study compared and characterized the tense and agreement productivity of boys with fragile X syndrome (FXS), children with developmental language disorder (DLD), and children with typical development (TD) matched on mean length of utterance. Method Twenty-two boys with FXS (M age = 12.22 years), 19 children with DLD (M age = 4.81 years), and 20 children with TD (M age = 3.23 years) produced language samples that were coded for th ...[more]

PMID: 32282263

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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

Comparing Tense and Agreement Productivity in Boys With Fragile X Syndrome, Children With Developmental Language Disorder, and Children With Typical Development.

Publications

Comparing Tense and Agreement Productivity in Boys With Fragile X Syndrome, Children With Developmental Language Disorder, and Children With Typical Development.

Similar Datasets

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