EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome

Introduction: Fragile X syndrome (FXS) is a genetic disorder caused by a mutation of the fragile X mental retardation 1 gene (FMR1). FXS is associated with neurophysiological abnormalities, including cortical hyperexcitability. Alterations in electroencephalogram (EEG) resting-state power spectral d...

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Autores principales: Mélodie Proteau-Lemieux, Inga Sophia Knoth, Kristian Agbogba, Valérie Côté, Hazel Maridith Barlahan Biag, Angela John Thurman, Charles-Olivier Martin, Anne-Marie Bélanger, Cory Rosenfelt, Flora Tassone, Leonard J. Abbeduto, Sébastien Jacquemont, Randi Hagerman, François Bolduc, David Hessl, Andrea Schneider, Sarah Lippé
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Publicado: Frontiers Media S.A. 2021
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Acceso en línea:https://doaj.org/article/d93ca7bdf2524fd8a1e7d8324ddc4651
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spelling oai:doaj.org-article:d93ca7bdf2524fd8a1e7d8324ddc46512021-11-11T09:27:43ZEEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome1664-064010.3389/fpsyt.2021.716707https://doaj.org/article/d93ca7bdf2524fd8a1e7d8324ddc46512021-11-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fpsyt.2021.716707/fullhttps://doaj.org/toc/1664-0640Introduction: Fragile X syndrome (FXS) is a genetic disorder caused by a mutation of the fragile X mental retardation 1 gene (FMR1). FXS is associated with neurophysiological abnormalities, including cortical hyperexcitability. Alterations in electroencephalogram (EEG) resting-state power spectral density (PSD) are well-defined in FXS and were found to be linked to neurodevelopmental delays. Whether non-linear dynamics of the brain signal are also altered remains to be studied.Methods: In this study, resting-state EEG power, including alpha peak frequency (APF) and theta/beta ratio (TBR), as well as signal complexity using multi-scale entropy (MSE) were compared between 26 FXS participants (ages 5–28 years), and 7 neurotypical (NT) controls with a similar age distribution. Subsequently a replication study was carried out, comparing our cohort to 19 FXS participants independently recorded at a different site.Results: PSD results confirmed the increased gamma, decreased alpha power and APF in FXS participants compared to NT controls. No alterations in TBR were found. Importantly, results revealed reduced signal complexity in FXS participants, specifically in higher scales, suggesting that altered signal complexity is sensitive to brain alterations in this population. The replication study mostly confirmed these results and suggested critical points of stagnation in the neurodevelopmental curve of FXS.Conclusion: Signal complexity is a powerful feature that can be added to the electrophysiological biomarkers of brain maturation in FXS.Mélodie Proteau-LemieuxMélodie Proteau-LemieuxInga Sophia KnothKristian AgbogbaValérie CôtéHazel Maridith Barlahan BiagAngela John ThurmanCharles-Olivier MartinAnne-Marie BélangerCory RosenfeltFlora TassoneFlora TassoneLeonard J. AbbedutoLeonard J. AbbedutoSébastien JacquemontSébastien JacquemontRandi HagermanFrançois BolducDavid HesslDavid HesslAndrea SchneiderAndrea SchneiderSarah LippéSarah LippéFrontiers Media S.A.articlefragile X syndromehyperexcitabilityEEG resting-statesignal complexitymultiscale entropyalpha peak frequencyPsychiatryRC435-571ENFrontiers in Psychiatry, Vol 12 (2021)
institution DOAJ
collection DOAJ
language EN
topic fragile X syndrome
hyperexcitability
EEG resting-state
signal complexity
multiscale entropy
alpha peak frequency
Psychiatry
RC435-571
spellingShingle fragile X syndrome
hyperexcitability
EEG resting-state
signal complexity
multiscale entropy
alpha peak frequency
Psychiatry
RC435-571
Mélodie Proteau-Lemieux
Mélodie Proteau-Lemieux
Inga Sophia Knoth
Kristian Agbogba
Valérie Côté
Hazel Maridith Barlahan Biag
Angela John Thurman
Charles-Olivier Martin
Anne-Marie Bélanger
Cory Rosenfelt
Flora Tassone
Flora Tassone
Leonard J. Abbeduto
Leonard J. Abbeduto
Sébastien Jacquemont
Sébastien Jacquemont
Randi Hagerman
François Bolduc
David Hessl
David Hessl
Andrea Schneider
Andrea Schneider
Sarah Lippé
Sarah Lippé
EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome
description Introduction: Fragile X syndrome (FXS) is a genetic disorder caused by a mutation of the fragile X mental retardation 1 gene (FMR1). FXS is associated with neurophysiological abnormalities, including cortical hyperexcitability. Alterations in electroencephalogram (EEG) resting-state power spectral density (PSD) are well-defined in FXS and were found to be linked to neurodevelopmental delays. Whether non-linear dynamics of the brain signal are also altered remains to be studied.Methods: In this study, resting-state EEG power, including alpha peak frequency (APF) and theta/beta ratio (TBR), as well as signal complexity using multi-scale entropy (MSE) were compared between 26 FXS participants (ages 5–28 years), and 7 neurotypical (NT) controls with a similar age distribution. Subsequently a replication study was carried out, comparing our cohort to 19 FXS participants independently recorded at a different site.Results: PSD results confirmed the increased gamma, decreased alpha power and APF in FXS participants compared to NT controls. No alterations in TBR were found. Importantly, results revealed reduced signal complexity in FXS participants, specifically in higher scales, suggesting that altered signal complexity is sensitive to brain alterations in this population. The replication study mostly confirmed these results and suggested critical points of stagnation in the neurodevelopmental curve of FXS.Conclusion: Signal complexity is a powerful feature that can be added to the electrophysiological biomarkers of brain maturation in FXS.
format article
author Mélodie Proteau-Lemieux
Mélodie Proteau-Lemieux
Inga Sophia Knoth
Kristian Agbogba
Valérie Côté
Hazel Maridith Barlahan Biag
Angela John Thurman
Charles-Olivier Martin
Anne-Marie Bélanger
Cory Rosenfelt
Flora Tassone
Flora Tassone
Leonard J. Abbeduto
Leonard J. Abbeduto
Sébastien Jacquemont
Sébastien Jacquemont
Randi Hagerman
François Bolduc
David Hessl
David Hessl
Andrea Schneider
Andrea Schneider
Sarah Lippé
Sarah Lippé
author_facet Mélodie Proteau-Lemieux
Mélodie Proteau-Lemieux
Inga Sophia Knoth
Kristian Agbogba
Valérie Côté
Hazel Maridith Barlahan Biag
Angela John Thurman
Charles-Olivier Martin
Anne-Marie Bélanger
Cory Rosenfelt
Flora Tassone
Flora Tassone
Leonard J. Abbeduto
Leonard J. Abbeduto
Sébastien Jacquemont
Sébastien Jacquemont
Randi Hagerman
François Bolduc
David Hessl
David Hessl
Andrea Schneider
Andrea Schneider
Sarah Lippé
Sarah Lippé
author_sort Mélodie Proteau-Lemieux
title EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome
title_short EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome
title_full EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome
title_fullStr EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome
title_full_unstemmed EEG Signal Complexity Is Reduced During Resting-State in Fragile X Syndrome
title_sort eeg signal complexity is reduced during resting-state in fragile x syndrome
publisher Frontiers Media S.A.
publishDate 2021
url https://doaj.org/article/d93ca7bdf2524fd8a1e7d8324ddc4651
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