Cargando…
Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome
Sensory hypersensitivities are common, clinically distressing features of Fragile X Syndrome (FXS). Preclinical evidence suggests this abnormality may result from synaptic hyper-excitability in sensory systems. This model predicts reduced sensory habituation to repeated stimulus presentation. Fourte...
Autores principales: | , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nature Publishing Group
2016
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4872406/ https://www.ncbi.nlm.nih.gov/pubmed/27093069 http://dx.doi.org/10.1038/tp.2016.48 |
_version_ | 1782432720565043200 |
---|---|
author | Ethridge, L E White, S P Mosconi, M W Wang, J Byerly, M J Sweeney, J A |
author_facet | Ethridge, L E White, S P Mosconi, M W Wang, J Byerly, M J Sweeney, J A |
author_sort | Ethridge, L E |
collection | PubMed |
description | Sensory hypersensitivities are common, clinically distressing features of Fragile X Syndrome (FXS). Preclinical evidence suggests this abnormality may result from synaptic hyper-excitability in sensory systems. This model predicts reduced sensory habituation to repeated stimulus presentation. Fourteen adolescents and adults with FXS and 15 age-matched controls participated in a modified auditory gating task using trains of 4 identical tones during dense array electroencephalography (EEG). Event-related potential and single trial time–frequency analyses revealed decreased habituation of the N1 event-related potential response in FXS, and increased gamma power coupled with decreases in gamma phase-locking during the early-stimulus registration period. EEG abnormalities in FXS were associated with parent reports of heightened sensory sensitivities and social communication deficits. Reduced habituation and altered gamma power and phase-locking to auditory cues demonstrated here in FXS patients parallels preclinical findings with Fmr1 KO mice. Thus, the EEG abnormalities seen in FXS patients support the model of neocortical hyper-excitability in FXS, and may provide useful translational biomarkers for evaluating novel treatment strategies targeting its neural substrate. |
format | Online Article Text |
id | pubmed-4872406 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2016 |
publisher | Nature Publishing Group |
record_format | MEDLINE/PubMed |
spelling | pubmed-48724062016-05-26 Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome Ethridge, L E White, S P Mosconi, M W Wang, J Byerly, M J Sweeney, J A Transl Psychiatry Original Article Sensory hypersensitivities are common, clinically distressing features of Fragile X Syndrome (FXS). Preclinical evidence suggests this abnormality may result from synaptic hyper-excitability in sensory systems. This model predicts reduced sensory habituation to repeated stimulus presentation. Fourteen adolescents and adults with FXS and 15 age-matched controls participated in a modified auditory gating task using trains of 4 identical tones during dense array electroencephalography (EEG). Event-related potential and single trial time–frequency analyses revealed decreased habituation of the N1 event-related potential response in FXS, and increased gamma power coupled with decreases in gamma phase-locking during the early-stimulus registration period. EEG abnormalities in FXS were associated with parent reports of heightened sensory sensitivities and social communication deficits. Reduced habituation and altered gamma power and phase-locking to auditory cues demonstrated here in FXS patients parallels preclinical findings with Fmr1 KO mice. Thus, the EEG abnormalities seen in FXS patients support the model of neocortical hyper-excitability in FXS, and may provide useful translational biomarkers for evaluating novel treatment strategies targeting its neural substrate. Nature Publishing Group 2016-04 2016-04-19 /pmc/articles/PMC4872406/ /pubmed/27093069 http://dx.doi.org/10.1038/tp.2016.48 Text en Copyright © 2016 Macmillan Publishers Limited http://creativecommons.org/licenses/by/4.0/ This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third party material in this article are included in the article's Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/ |
spellingShingle | Original Article Ethridge, L E White, S P Mosconi, M W Wang, J Byerly, M J Sweeney, J A Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome |
title | Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome |
title_full | Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome |
title_fullStr | Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome |
title_full_unstemmed | Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome |
title_short | Reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in Fragile X Syndrome |
title_sort | reduced habituation of auditory evoked potentials indicate cortical hyper-excitability in fragile x syndrome |
topic | Original Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4872406/ https://www.ncbi.nlm.nih.gov/pubmed/27093069 http://dx.doi.org/10.1038/tp.2016.48 |
work_keys_str_mv | AT ethridgele reducedhabituationofauditoryevokedpotentialsindicatecorticalhyperexcitabilityinfragilexsyndrome AT whitesp reducedhabituationofauditoryevokedpotentialsindicatecorticalhyperexcitabilityinfragilexsyndrome AT mosconimw reducedhabituationofauditoryevokedpotentialsindicatecorticalhyperexcitabilityinfragilexsyndrome AT wangj reducedhabituationofauditoryevokedpotentialsindicatecorticalhyperexcitabilityinfragilexsyndrome AT byerlymj reducedhabituationofauditoryevokedpotentialsindicatecorticalhyperexcitabilityinfragilexsyndrome AT sweeneyja reducedhabituationofauditoryevokedpotentialsindicatecorticalhyperexcitabilityinfragilexsyndrome |