[article]
| Titre : |
Neural synchronization deficits linked to cortical hyper-excitability and auditory hypersensitivity in fragile X syndrome |
| Type de document : |
texte imprimé |
| Auteurs : |
Lauren E ETHRIDGE, Auteur ; Stormi P. WHITE, Auteur ; Matthew W. MOSCONI, Auteur ; Jing WANG, Auteur ; Ernest V. PEDAPATI, Auteur ; Craig ERICKSON, Auteur ; Matthew J. BYERLY, Auteur ; John A. SWEENEY, Auteur |
| Article en page(s) : |
22p. |
| Langues : |
Anglais (eng) |
| Mots-clés : |
Chirp Eeg Fragile X syndrome Gamma Sensory |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Studies in the fmr1 KO mouse demonstrate hyper-excitability and increased high-frequency neuronal activity in sensory cortex. These abnormalities may contribute to prominent and distressing sensory hypersensitivities in patients with fragile X syndrome (FXS). The current study investigated functional properties of auditory cortex using a sensory entrainment task in FXS. METHODS: EEG recordings were obtained from 17 adolescents and adults with FXS and 17 age- and sex-matched healthy controls. Participants heard an auditory chirp stimulus generated using a 1000-Hz tone that was amplitude modulated by a sinusoid linearly increasing in frequency from 0-100 Hz over 2 s. RESULTS: Single trial time-frequency analyses revealed decreased gamma band phase-locking to the chirp stimulus in FXS, which was strongly coupled with broadband increases in gamma power. Abnormalities in gamma phase-locking and power were also associated with theta-gamma amplitude-amplitude coupling during the pre-stimulus period and with parent reports of heightened sensory sensitivities and social communication deficits. CONCLUSIONS: This represents the first demonstration of neural entrainment alterations in FXS patients and suggests that fast-spiking interneurons regulating synchronous high-frequency neural activity have reduced functionality. This reduced ability to synchronize high-frequency neural activity was related to the total power of background gamma band activity. These observations extend findings from fmr1 KO models of FXS, characterize a core pathophysiological aspect of FXS, and may provide a translational biomarker strategy for evaluating promising therapeutics. |
| En ligne : |
http://dx.doi.org/10.1186/s13229-017-0140-1 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=330 |
in Molecular Autism > 8 (2017) . - 22p.
[article] Neural synchronization deficits linked to cortical hyper-excitability and auditory hypersensitivity in fragile X syndrome [texte imprimé] / Lauren E ETHRIDGE, Auteur ; Stormi P. WHITE, Auteur ; Matthew W. MOSCONI, Auteur ; Jing WANG, Auteur ; Ernest V. PEDAPATI, Auteur ; Craig ERICKSON, Auteur ; Matthew J. BYERLY, Auteur ; John A. SWEENEY, Auteur . - 22p. Langues : Anglais ( eng) in Molecular Autism > 8 (2017) . - 22p.
| Mots-clés : |
Chirp Eeg Fragile X syndrome Gamma Sensory |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Studies in the fmr1 KO mouse demonstrate hyper-excitability and increased high-frequency neuronal activity in sensory cortex. These abnormalities may contribute to prominent and distressing sensory hypersensitivities in patients with fragile X syndrome (FXS). The current study investigated functional properties of auditory cortex using a sensory entrainment task in FXS. METHODS: EEG recordings were obtained from 17 adolescents and adults with FXS and 17 age- and sex-matched healthy controls. Participants heard an auditory chirp stimulus generated using a 1000-Hz tone that was amplitude modulated by a sinusoid linearly increasing in frequency from 0-100 Hz over 2 s. RESULTS: Single trial time-frequency analyses revealed decreased gamma band phase-locking to the chirp stimulus in FXS, which was strongly coupled with broadband increases in gamma power. Abnormalities in gamma phase-locking and power were also associated with theta-gamma amplitude-amplitude coupling during the pre-stimulus period and with parent reports of heightened sensory sensitivities and social communication deficits. CONCLUSIONS: This represents the first demonstration of neural entrainment alterations in FXS patients and suggests that fast-spiking interneurons regulating synchronous high-frequency neural activity have reduced functionality. This reduced ability to synchronize high-frequency neural activity was related to the total power of background gamma band activity. These observations extend findings from fmr1 KO models of FXS, characterize a core pathophysiological aspect of FXS, and may provide a translational biomarker strategy for evaluating promising therapeutics. |
| En ligne : |
http://dx.doi.org/10.1186/s13229-017-0140-1 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=330 |
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Neural synchronization deficits linked to cortical hyper-excitability and auditory hypersensitivity in fragile X syndrome in Molecular Autism, 8 (2017)
