EEG as a translational biomarker and outcome measure in fragile X syndrome
Abstract 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...
| Main Authors: | , , |
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| Format: | Article |
| Language: | English |
| Published: |
Nature Publishing Group
2022-01-01
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| Series: | Translational Psychiatry |
| Online Access: | https://doi.org/10.1038/s41398-022-01796-2 |
| _version_ | 1819280398539030528 |
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| author | Aisling Kenny Damien Wright Andrew C. Stanfield |
| author_facet | Aisling Kenny Damien Wright Andrew C. Stanfield |
| author_sort | Aisling Kenny |
| collection | DOAJ |
| description | Abstract 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. |
| first_indexed | 2024-12-24T00:43:10Z |
| format | Article |
| id | doaj.art-682bb96ce5ee44bd92a91d6b5ed50187 |
| institution | Directory Open Access Journal |
| issn | 2158-3188 |
| language | English |
| last_indexed | 2024-12-24T00:43:10Z |
| publishDate | 2022-01-01 |
| publisher | Nature Publishing Group |
| record_format | Article |
| series | Translational Psychiatry |
| spelling | doaj.art-682bb96ce5ee44bd92a91d6b5ed501872022-12-21T17:23:52ZengNature Publishing GroupTranslational Psychiatry2158-31882022-01-0112111210.1038/s41398-022-01796-2EEG as a translational biomarker and outcome measure in fragile X syndromeAisling Kenny0Damien Wright1Andrew C. Stanfield2Patrick Wild Centre, Division of Psychiatry, Kennedy Tower, Royal Edinburgh Hospital, University of EdinburghPatrick Wild Centre, Division of Psychiatry, Kennedy Tower, Royal Edinburgh Hospital, University of EdinburghPatrick Wild Centre, Division of Psychiatry, Kennedy Tower, Royal Edinburgh Hospital, University of EdinburghAbstract 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.https://doi.org/10.1038/s41398-022-01796-2 |
| spellingShingle | Aisling Kenny Damien Wright Andrew C. Stanfield EEG as a translational biomarker and outcome measure in fragile X syndrome Translational Psychiatry |
| 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 |
| url | https://doi.org/10.1038/s41398-022-01796-2 |
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