Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA

Detalhes bibliográficos
Autor(a) principal: Leal, A
Data de Publicação: 2016
Outros Autores: Vieira, JP, Lopes, R, Nunes, R, Gonçalves, S, Lopes da Silva, F, Figueiredo, P
Tipo de documento: Artigo
Idioma: eng
Título da fonte: Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
Texto Completo: http://hdl.handle.net/10400.17/2475
Resumo: Childhood absence epilepsy (CAE) is a syndrome with well-defined electroclinical features but unknown pathological basis. An increased thalamic tonic GABA inhibition has recently been discovered on animal models (Cope et al., 2009), but its relevance for human CAE is unproven. METHODS: We studied an 11-year-old boy, presenting the typical clinical features of CAE, but spike-wave discharges (SWD) restricted to one hemisphere. RESULTS: High-resolution EEG failed to demonstrate independent contralateral hemisphere epileptic activity. Consistently, simultaneous EEG-fMRI revealed the typical thalamic BOLD activation, associated with caudate and default mode network deactivation, but restricted to the hemisphere with SWD. Cortical BOLD activations were localized on the ipsilateral pars transverse. Magnetic resonance spectroscopy, using MEGA-PRESS, showed that the GABA/creatine ratio was 2.6 times higher in the hemisphere with SWD than in the unaffected one, reflecting a higher GABA concentration. Similar comparisons for the patient's occipital cortex and thalamus of a healthy volunteer yielded asymmetries below 25%. SIGNIFICANCE: In a clinical case of CAE with EEG and fMRI-BOLD manifestations restricted to one hemisphere, we found an associated increase in thalamic GABA concentration consistent with a role for this abnormality in human CAE.
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spelling Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABAChildhood absence epilepsyGABAThalamusChildHDE NEU PEDChildhood absence epilepsy (CAE) is a syndrome with well-defined electroclinical features but unknown pathological basis. An increased thalamic tonic GABA inhibition has recently been discovered on animal models (Cope et al., 2009), but its relevance for human CAE is unproven. METHODS: We studied an 11-year-old boy, presenting the typical clinical features of CAE, but spike-wave discharges (SWD) restricted to one hemisphere. RESULTS: High-resolution EEG failed to demonstrate independent contralateral hemisphere epileptic activity. Consistently, simultaneous EEG-fMRI revealed the typical thalamic BOLD activation, associated with caudate and default mode network deactivation, but restricted to the hemisphere with SWD. Cortical BOLD activations were localized on the ipsilateral pars transverse. Magnetic resonance spectroscopy, using MEGA-PRESS, showed that the GABA/creatine ratio was 2.6 times higher in the hemisphere with SWD than in the unaffected one, reflecting a higher GABA concentration. Similar comparisons for the patient's occipital cortex and thalamus of a healthy volunteer yielded asymmetries below 25%. SIGNIFICANCE: In a clinical case of CAE with EEG and fMRI-BOLD manifestations restricted to one hemisphere, we found an associated increase in thalamic GABA concentration consistent with a role for this abnormality in human CAE.Elsevier Inc.Repositório do Centro Hospitalar Universitário de Lisboa Central, EPELeal, AVieira, JPLopes, RNunes, RGonçalves, SLopes da Silva, FFigueiredo, P2016-05-10T13:26:58Z20162016-01-01T00:00:00Zinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfhttp://hdl.handle.net/10400.17/2475engEpilepsy Behav Case Rep. 2016 Apr 6;5:57-6510.1016/j.ebcr.2016.03.004info:eu-repo/semantics/openAccessreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAP2023-03-10T09:37:16Zoai:repositorio.chlc.min-saude.pt:10400.17/2475Portal AgregadorONGhttps://www.rcaap.pt/oai/openaireopendoar:71602024-03-19T17:19:49.489189Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãofalse
dc.title.none.fl_str_mv Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
title Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
spellingShingle Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
Leal, A
Childhood absence epilepsy
GABA
Thalamus
Child
HDE NEU PED
title_short Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
title_full Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
title_fullStr Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
title_full_unstemmed Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
title_sort Dynamics of Epileptic Activity in a Peculiar Case of Childhood Absence Epilepsy and Correlation with Thalamic Levels of GABA
author Leal, A
author_facet Leal, A
Vieira, JP
Lopes, R
Nunes, R
Gonçalves, S
Lopes da Silva, F
Figueiredo, P
author_role author
author2 Vieira, JP
Lopes, R
Nunes, R
Gonçalves, S
Lopes da Silva, F
Figueiredo, P
author2_role author
author
author
author
author
author
dc.contributor.none.fl_str_mv Repositório do Centro Hospitalar Universitário de Lisboa Central, EPE
dc.contributor.author.fl_str_mv Leal, A
Vieira, JP
Lopes, R
Nunes, R
Gonçalves, S
Lopes da Silva, F
Figueiredo, P
dc.subject.por.fl_str_mv Childhood absence epilepsy
GABA
Thalamus
Child
HDE NEU PED
topic Childhood absence epilepsy
GABA
Thalamus
Child
HDE NEU PED
description Childhood absence epilepsy (CAE) is a syndrome with well-defined electroclinical features but unknown pathological basis. An increased thalamic tonic GABA inhibition has recently been discovered on animal models (Cope et al., 2009), but its relevance for human CAE is unproven. METHODS: We studied an 11-year-old boy, presenting the typical clinical features of CAE, but spike-wave discharges (SWD) restricted to one hemisphere. RESULTS: High-resolution EEG failed to demonstrate independent contralateral hemisphere epileptic activity. Consistently, simultaneous EEG-fMRI revealed the typical thalamic BOLD activation, associated with caudate and default mode network deactivation, but restricted to the hemisphere with SWD. Cortical BOLD activations were localized on the ipsilateral pars transverse. Magnetic resonance spectroscopy, using MEGA-PRESS, showed that the GABA/creatine ratio was 2.6 times higher in the hemisphere with SWD than in the unaffected one, reflecting a higher GABA concentration. Similar comparisons for the patient's occipital cortex and thalamus of a healthy volunteer yielded asymmetries below 25%. SIGNIFICANCE: In a clinical case of CAE with EEG and fMRI-BOLD manifestations restricted to one hemisphere, we found an associated increase in thalamic GABA concentration consistent with a role for this abnormality in human CAE.
publishDate 2016
dc.date.none.fl_str_mv 2016-05-10T13:26:58Z
2016
2016-01-01T00:00:00Z
dc.type.status.fl_str_mv info:eu-repo/semantics/publishedVersion
dc.type.driver.fl_str_mv info:eu-repo/semantics/article
format article
status_str publishedVersion
dc.identifier.uri.fl_str_mv http://hdl.handle.net/10400.17/2475
url http://hdl.handle.net/10400.17/2475
dc.language.iso.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv Epilepsy Behav Case Rep. 2016 Apr 6;5:57-65
10.1016/j.ebcr.2016.03.004
dc.rights.driver.fl_str_mv info:eu-repo/semantics/openAccess
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
dc.publisher.none.fl_str_mv Elsevier Inc.
publisher.none.fl_str_mv Elsevier Inc.
dc.source.none.fl_str_mv reponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
instacron:RCAAP
instname_str Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
instacron_str RCAAP
institution RCAAP
reponame_str Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
collection Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
repository.name.fl_str_mv Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
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