Increased aperiodic gamma power in young boys with Fragile X Syndrome is associated with better language ability / C. L. WILKINSON in Molecular Autism, 12 (2021)  

Public ISBD [article]  inMolecular Autism > 12 (2021) . - 17 p. Titre : Increased aperiodic gamma power in young boys with Fragile X Syndrome is associated with better language ability Type de document : Texte imprimé et/ou numérique Auteurs : C. L. WILKINSON, Auteur ; C. A. NELSON, Auteur Article en page(s) : 17 p. Langues : Anglais (eng) Mots-clés : Brain/physiopathology  Child  Child Language  Child, Preschool  Electroencephalography  Fragile X Mental Retardation Protein/genetics  Fragile X Syndrome/genetics/physiopathology/psychology  Humans  Male  Biomarker  E:I ratio  Fragile X Syndrome  Gamma  Language  Outcome measures Index. décimale : PER Périodiques Résumé : BACKGROUND: The 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. METHODS: Resting-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. RESULTS: FXS 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. LIMITATIONS: The 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. CONCLUSIONS: Resting-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. En ligne : http://dx.doi.org/10.1186/s13229-021-00425-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=459 [article] Increased aperiodic gamma power in young boys with Fragile X Syndrome is associated with better language ability [Texte imprimé et/ou numérique] / C. L. WILKINSON, Auteur ; C. A. NELSON, Auteur . - 17 p. Langues : Anglais (eng) in Molecular Autism > 12 (2021) . - 17 p. Mots-clés : Brain/physiopathology  Child  Child Language  Child, Preschool  Electroencephalography  Fragile X Mental Retardation Protein/genetics  Fragile X Syndrome/genetics/physiopathology/psychology  Humans  Male  Biomarker  E:I ratio  Fragile X Syndrome  Gamma  Language  Outcome measures Index. décimale : PER Périodiques Résumé : BACKGROUND: The 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. METHODS: Resting-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. RESULTS: FXS 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. LIMITATIONS: The 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. CONCLUSIONS: Resting-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. En ligne : http://dx.doi.org/10.1186/s13229-021-00425-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=459

Reduced frontal gamma power at 24 months is associated with better expressive language in toddlers at risk for autism / C. L. WILKINSON in Autism Research, 12-8 (August 2019)  

Public ISBD [article]  inAutism Research > 12-8 (August 2019) . - p.1211-1224 Titre : Reduced frontal gamma power at 24 months is associated with better expressive language in toddlers at risk for autism Type de document : Texte imprimé et/ou numérique Auteurs : C. L. WILKINSON, Auteur ; A. R. LEVIN, Auteur ; L. J. GABARD-DURNAM, Auteur ; Helen TAGER-FLUSBERG, Auteur ; C. A. NELSON, Auteur Article en page(s) : p.1211-1224 Langues : Anglais (eng) Mots-clés : children  cognitive neuroscience  electroencephalography (EEG)  infants  language Index. décimale : PER Périodiques Résumé : Frontal gamma power has been associated with early language development in typically developing toddlers, and gamma band abnormalities have been observed in individuals with autism spectrum disorder (ASD), as well as high-risk infant siblings (those having an older sibling with ASD), as early as 6 months of age. The current study investigated differences in baseline frontal gamma power and its association with language development in toddlers at high versus low familial risk for autism. Electroencephalography recordings as well as cognitive and behavioral assessments were acquired at 24 months as part of prospective, longitudinal study of infant siblings of children with and without autism. Diagnosis of autism was determined at 24-36 months, and data were analyzed across three outcome groups-low-risk without ASD (n = 43), high-risk without ASD (n = 42), and high-risk with ASD (n = 16). High-risk toddlers without ASD had reduced baseline frontal gamma power (30-50 Hz) compared to low-risk toddlers. Among high-risk toddlers increased frontal gamma was only marginally associated with ASD diagnosis (P = 0.06), but significantly associated with reduced expressive language ability (P = 0.007). No association between gamma power and language was present in the low-risk group. These findings suggest that differences in gamma oscillations in high-risk toddlers may represent compensatory mechanisms associated with improved developmental outcomes. Autism Res 2019, 12: 1211-1224. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: This study looked at differences in neural activity in the gamma range and its association with language in toddlers with and without increased risk for ASD. At 2 years of age, gamma power was lower in high-risk toddlers without ASD compared to a low-risk comparison group. Among high-risk toddlers both with and without later ASD, reduced gamma power was also associated with better language outcomes, suggesting that gamma power may be a marker of language development in high-risk children. En ligne : http://dx.doi.org/10.1002/aur.2131 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=405 [article] Reduced frontal gamma power at 24 months is associated with better expressive language in toddlers at risk for autism [Texte imprimé et/ou numérique] / C. L. WILKINSON, Auteur ; A. R. LEVIN, Auteur ; L. J. GABARD-DURNAM, Auteur ; Helen TAGER-FLUSBERG, Auteur ; C. A. NELSON, Auteur . - p.1211-1224. Langues : Anglais (eng) in Autism Research > 12-8 (August 2019) . - p.1211-1224 Mots-clés : children  cognitive neuroscience  electroencephalography (EEG)  infants  language Index. décimale : PER Périodiques Résumé : Frontal gamma power has been associated with early language development in typically developing toddlers, and gamma band abnormalities have been observed in individuals with autism spectrum disorder (ASD), as well as high-risk infant siblings (those having an older sibling with ASD), as early as 6 months of age. The current study investigated differences in baseline frontal gamma power and its association with language development in toddlers at high versus low familial risk for autism. Electroencephalography recordings as well as cognitive and behavioral assessments were acquired at 24 months as part of prospective, longitudinal study of infant siblings of children with and without autism. Diagnosis of autism was determined at 24-36 months, and data were analyzed across three outcome groups-low-risk without ASD (n = 43), high-risk without ASD (n = 42), and high-risk with ASD (n = 16). High-risk toddlers without ASD had reduced baseline frontal gamma power (30-50 Hz) compared to low-risk toddlers. Among high-risk toddlers increased frontal gamma was only marginally associated with ASD diagnosis (P = 0.06), but significantly associated with reduced expressive language ability (P = 0.007). No association between gamma power and language was present in the low-risk group. These findings suggest that differences in gamma oscillations in high-risk toddlers may represent compensatory mechanisms associated with improved developmental outcomes. Autism Res 2019, 12: 1211-1224. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: This study looked at differences in neural activity in the gamma range and its association with language in toddlers with and without increased risk for ASD. At 2 years of age, gamma power was lower in high-risk toddlers without ASD compared to a low-risk comparison group. Among high-risk toddlers both with and without later ASD, reduced gamma power was also associated with better language outcomes, suggesting that gamma power may be a marker of language development in high-risk children. En ligne : http://dx.doi.org/10.1002/aur.2131 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=405

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