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EEG as a translational biomarker and outcome measure in fragile X syndrome

Targeted treatments for fragile X syndrome (FXS) have frequently failed to show efficacy in clinical testing, despite success at the preclinical stages. This has highlighted the need for more effective translational outcome measures. EEG differences observed in FXS, including exaggerated N1 ERP ampl...

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Detalles Bibliográficos
Autores principales: Kenny, Aisling, Wright, Damien, Stanfield, Andrew C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8786970/
https://www.ncbi.nlm.nih.gov/pubmed/35075104
http://dx.doi.org/10.1038/s41398-022-01796-2
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author Kenny, Aisling
Wright, Damien
Stanfield, Andrew C.
author_facet Kenny, Aisling
Wright, Damien
Stanfield, Andrew C.
author_sort Kenny, Aisling
collection PubMed
description Targeted treatments for fragile X syndrome (FXS) have frequently failed to show efficacy in clinical testing, despite success at the preclinical stages. This has highlighted the need for more effective translational outcome measures. EEG differences observed in FXS, including exaggerated N1 ERP amplitudes, increased resting gamma power and reduced gamma phase-locking in the sensory cortices, have been suggested as potential biomarkers of the syndrome. These abnormalities are thought to reflect cortical hyper excitability resulting from an excitatory (glutamate) and inhibitory (GABAergic) imbalance in FXS, which has been the target of several pharmaceutical remediation studies. EEG differences observed in humans also show similarities to those seen in laboratory models of FXS, which may allow for greater translational equivalence and better predict clinical success of putative therapeutics. There is some evidence from clinical trials showing that treatment related changes in EEG may be associated with clinical improvements, but these require replication and extension to other medications. Although the use of EEG characteristics as biomarkers is still in the early phases, and further research is needed to establish its utility in clinical trials, the current research is promising and signals the emergence of an effective translational biomarker.
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spelling pubmed-87869702022-02-07 EEG as a translational biomarker and outcome measure in fragile X syndrome Kenny, Aisling Wright, Damien Stanfield, Andrew C. Transl Psychiatry Review Article Targeted treatments for fragile X syndrome (FXS) have frequently failed to show efficacy in clinical testing, despite success at the preclinical stages. This has highlighted the need for more effective translational outcome measures. EEG differences observed in FXS, including exaggerated N1 ERP amplitudes, increased resting gamma power and reduced gamma phase-locking in the sensory cortices, have been suggested as potential biomarkers of the syndrome. These abnormalities are thought to reflect cortical hyper excitability resulting from an excitatory (glutamate) and inhibitory (GABAergic) imbalance in FXS, which has been the target of several pharmaceutical remediation studies. EEG differences observed in humans also show similarities to those seen in laboratory models of FXS, which may allow for greater translational equivalence and better predict clinical success of putative therapeutics. There is some evidence from clinical trials showing that treatment related changes in EEG may be associated with clinical improvements, but these require replication and extension to other medications. Although the use of EEG characteristics as biomarkers is still in the early phases, and further research is needed to establish its utility in clinical trials, the current research is promising and signals the emergence of an effective translational biomarker. Nature Publishing Group UK 2022-01-24 /pmc/articles/PMC8786970/ /pubmed/35075104 http://dx.doi.org/10.1038/s41398-022-01796-2 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Review Article
Kenny, Aisling
Wright, Damien
Stanfield, Andrew C.
EEG as a translational biomarker and outcome measure in fragile X syndrome
title EEG as a translational biomarker and outcome measure in fragile X syndrome
title_full EEG as a translational biomarker and outcome measure in fragile X syndrome
title_fullStr EEG as a translational biomarker and outcome measure in fragile X syndrome
title_full_unstemmed EEG as a translational biomarker and outcome measure in fragile X syndrome
title_short EEG as a translational biomarker and outcome measure in fragile X syndrome
title_sort eeg as a translational biomarker and outcome measure in fragile x syndrome
topic Review Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8786970/
https://www.ncbi.nlm.nih.gov/pubmed/35075104
http://dx.doi.org/10.1038/s41398-022-01796-2
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