CPEB4–CLOCK crosstalk during temporal lobe epilepsy

Objective: Posttranscriptional mechanisms are increasingly recognized as important contributors to the formation of hyperexcitable networks in epilepsy. Messenger RNA (mRNA) polyadenylation is a key regulatory mechanism governing protein expression by enhancing mRNA stability and translation. Previo...

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Autores: de Diego-Garcia, Laura, Brennan, Gary P, Auer, Theresa, Menendez-Mendez, Aida, Parras, Alberto, Martin-Gil, Alba, Mitra, Meghma, Ollà, Ivana, Villalba-Benito, Leticia, Gil, Beatriz, Alves, Mariana, Lau, Kelvin, Delanty, Norman, Beausang, Alan, Cryan, Jane, Brett, Francesca M., Farrell, Michael A., O'Brien, Donncha F., Mendez, Raúl, Carracedo-Rodríguez, Gonzalo, Henshall, David C, Lucas, José J, Engel, Tobias
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2023
País:España
Institución:Consejo Superior de Investigaciones Científicas (CSIC)
Repositorio:DIGITAL.CSIC. Repositorio Institucional del CSIC
OAI Identifier:oai:digital.csic.es:10261/346753
Acceso en línea:http://hdl.handle.net/10261/346753
Access Level:acceso abierto
Palabra clave:Circadian rhythm
CLOCK
CPEB4
Cytoplasmic polyadenylation
Epilepsy
Status epilepticus
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oai_identifier_str oai:digital.csic.es:10261/346753
network_acronym_str ES
network_name_str España
repository_id_str
dc.title.none.fl_str_mv CPEB4–CLOCK crosstalk during temporal lobe epilepsy
title CPEB4–CLOCK crosstalk during temporal lobe epilepsy
spellingShingle CPEB4–CLOCK crosstalk during temporal lobe epilepsy
de Diego-Garcia, Laura
Circadian rhythm
CLOCK
CPEB4
Cytoplasmic polyadenylation
Epilepsy
Status epilepticus
title_short CPEB4–CLOCK crosstalk during temporal lobe epilepsy
title_full CPEB4–CLOCK crosstalk during temporal lobe epilepsy
title_fullStr CPEB4–CLOCK crosstalk during temporal lobe epilepsy
title_full_unstemmed CPEB4–CLOCK crosstalk during temporal lobe epilepsy
title_sort CPEB4–CLOCK crosstalk during temporal lobe epilepsy
dc.creator.none.fl_str_mv de Diego-Garcia, Laura
Brennan, Gary P
Auer, Theresa
Menendez-Mendez, Aida
Parras, Alberto
Martin-Gil, Alba
Mitra, Meghma
Ollà, Ivana
Villalba-Benito, Leticia
Gil, Beatriz
Alves, Mariana
Lau, Kelvin
Delanty, Norman
Beausang, Alan
Cryan, Jane
Brett, Francesca M.
Farrell, Michael A.
O'Brien, Donncha F.
Mendez, Raúl
Carracedo-Rodríguez, Gonzalo
Henshall, David C
Lucas, José J
Engel, Tobias
author de Diego-Garcia, Laura
author_facet de Diego-Garcia, Laura
Brennan, Gary P
Auer, Theresa
Menendez-Mendez, Aida
Parras, Alberto
Martin-Gil, Alba
Mitra, Meghma
Ollà, Ivana
Villalba-Benito, Leticia
Gil, Beatriz
Alves, Mariana
Lau, Kelvin
Delanty, Norman
Beausang, Alan
Cryan, Jane
Brett, Francesca M.
Farrell, Michael A.
O'Brien, Donncha F.
Mendez, Raúl
Carracedo-Rodríguez, Gonzalo
Henshall, David C
Lucas, José J
Engel, Tobias
author_role author
author2 Brennan, Gary P
Auer, Theresa
Menendez-Mendez, Aida
Parras, Alberto
Martin-Gil, Alba
Mitra, Meghma
Ollà, Ivana
Villalba-Benito, Leticia
Gil, Beatriz
Alves, Mariana
Lau, Kelvin
Delanty, Norman
Beausang, Alan
Cryan, Jane
Brett, Francesca M.
Farrell, Michael A.
O'Brien, Donncha F.
Mendez, Raúl
Carracedo-Rodríguez, Gonzalo
Henshall, David C
Lucas, José J
Engel, Tobias
author2_role author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
dc.contributor.none.fl_str_mv European Commission
Irish Research Council
Comunidad de Madrid
Science Foundation Ireland
Ministerio de Economía y Competitividad (España)
Ministerio de Ciencia, Innovación y Universidades (España)
Consejo Superior de Investigaciones Científicas [https://ror.org/02gfc7t72]
dc.subject.none.fl_str_mv Circadian rhythm
CLOCK
CPEB4
Cytoplasmic polyadenylation
Epilepsy
Status epilepticus
topic Circadian rhythm
CLOCK
CPEB4
Cytoplasmic polyadenylation
Epilepsy
Status epilepticus
description Objective: Posttranscriptional mechanisms are increasingly recognized as important contributors to the formation of hyperexcitable networks in epilepsy. Messenger RNA (mRNA) polyadenylation is a key regulatory mechanism governing protein expression by enhancing mRNA stability and translation. Previous studies have shown large-scale changes in mRNA polyadenylation in the hippocampus of mice during epilepsy development. The cytoplasmic polyadenylation element-binding protein CPEB4 was found to drive epilepsy-induced poly(A) tail changes, and mice lacking CPEB4 develop a more severe seizure and epilepsy phenotype. The mechanisms controlling CPEB4 function and the downstream pathways that influence the recurrence of spontaneous seizures in epilepsy remain poorly understood. Methods: Status epilepticus was induced in wild-type and CPEB4-deficient male mice via an intra-amygdala microinjection of kainic acid. CLOCK binding to the CPEB4 promoter was analyzed via chromatin immunoprecipitation assay and melatonin levels via high-performance liquid chromatography in plasma. Results: Here, we show increased binding of CLOCK to recognition sites in the CPEB4 promoter region during status epilepticus in mice and increased Cpeb4 mRNA levels in N2A cells overexpressing CLOCK. Bioinformatic analysis of CPEB4-dependent genes undergoing changes in their poly(A) tail during epilepsy found that genes involved in the regulation of circadian rhythms are particularly enriched. Clock transcripts displayed a longer poly(A) tail length in the hippocampus of mice post-status epilepticus and during epilepsy. Moreover, CLOCK expression was increased in the hippocampus in mice post-status epilepticus and during epilepsy, and in resected hippocampus and cortex of patients with drug-resistant temporal lobe epilepsy. Furthermore, CPEB4 is required for CLOCK expression after status epilepticus, with lower levels in CPEB4-deficient compared to wild-type mice. Last, CPEB4-deficient mice showed altered circadian function, including altered melatonin blood levels and altered clustering of spontaneous seizures during the day. Significance: Our results reveal a new positive transcriptional–translational feedback loop involving CPEB4 and CLOCK, which may contribute to the regulation of the sleep–wake cycle during epilepsy.
publishDate 2023
dc.date.none.fl_str_mv 2023
2024
2024
2024
dc.type.none.fl_str_mv info:eu-repo/semantics/article
http://purl.org/coar/resource_type/c_6501
Publisher's version
info:eu-repo/semantics/publishedVersion
format article
status_str publishedVersion
dc.identifier.none.fl_str_mv http://hdl.handle.net/10261/346753
url http://hdl.handle.net/10261/346753
dc.language.none.fl_str_mv Inglés
language_invalid_str_mv Inglés
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info:eu-repo/grantAgreement/EC/H2020/766124
info:eu-repo/grantAgreement/EC/H2020/796600
info:eu-repo/grantAgreement/AEI/Plan Estatal de Investigación Científica y Técnica y de Innovación 2021-2023/PID2021-123141OB-I00
2020-T2/BMD-20180/ProgramadeAtraccióndeTalento-CM
http://dx.doi.org/10.1111/epi.17736

dc.rights.none.fl_str_mv info:eu-repo/semantics/openAccess
eu_rights_str_mv openAccess
dc.publisher.none.fl_str_mv John Wiley & Sons
publisher.none.fl_str_mv John Wiley & Sons
dc.source.none.fl_str_mv reponame:DIGITAL.CSIC. Repositorio Institucional del CSIC
instname:Consejo Superior de Investigaciones Científicas (CSIC)
instname_str Consejo Superior de Investigaciones Científicas (CSIC)
reponame_str DIGITAL.CSIC. Repositorio Institucional del CSIC
collection DIGITAL.CSIC. Repositorio Institucional del CSIC
repository.name.fl_str_mv
repository.mail.fl_str_mv
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spelling CPEB4–CLOCK crosstalk during temporal lobe epilepsyde Diego-Garcia, LauraBrennan, Gary PAuer, TheresaMenendez-Mendez, AidaParras, AlbertoMartin-Gil, AlbaMitra, MeghmaOllà, IvanaVillalba-Benito, LeticiaGil, BeatrizAlves, MarianaLau, KelvinDelanty, NormanBeausang, AlanCryan, JaneBrett, Francesca M.Farrell, Michael A.O'Brien, Donncha F.Mendez, RaúlCarracedo-Rodríguez, GonzaloHenshall, David CLucas, José JEngel, TobiasCircadian rhythmCLOCKCPEB4Cytoplasmic polyadenylationEpilepsyStatus epilepticusObjective: Posttranscriptional mechanisms are increasingly recognized as important contributors to the formation of hyperexcitable networks in epilepsy. Messenger RNA (mRNA) polyadenylation is a key regulatory mechanism governing protein expression by enhancing mRNA stability and translation. Previous studies have shown large-scale changes in mRNA polyadenylation in the hippocampus of mice during epilepsy development. The cytoplasmic polyadenylation element-binding protein CPEB4 was found to drive epilepsy-induced poly(A) tail changes, and mice lacking CPEB4 develop a more severe seizure and epilepsy phenotype. The mechanisms controlling CPEB4 function and the downstream pathways that influence the recurrence of spontaneous seizures in epilepsy remain poorly understood. Methods: Status epilepticus was induced in wild-type and CPEB4-deficient male mice via an intra-amygdala microinjection of kainic acid. CLOCK binding to the CPEB4 promoter was analyzed via chromatin immunoprecipitation assay and melatonin levels via high-performance liquid chromatography in plasma. Results: Here, we show increased binding of CLOCK to recognition sites in the CPEB4 promoter region during status epilepticus in mice and increased Cpeb4 mRNA levels in N2A cells overexpressing CLOCK. Bioinformatic analysis of CPEB4-dependent genes undergoing changes in their poly(A) tail during epilepsy found that genes involved in the regulation of circadian rhythms are particularly enriched. Clock transcripts displayed a longer poly(A) tail length in the hippocampus of mice post-status epilepticus and during epilepsy. Moreover, CLOCK expression was increased in the hippocampus in mice post-status epilepticus and during epilepsy, and in resected hippocampus and cortex of patients with drug-resistant temporal lobe epilepsy. Furthermore, CPEB4 is required for CLOCK expression after status epilepticus, with lower levels in CPEB4-deficient compared to wild-type mice. Last, CPEB4-deficient mice showed altered circadian function, including altered melatonin blood levels and altered clustering of spontaneous seizures during the day. Significance: Our results reveal a new positive transcriptional–translational feedback loop involving CPEB4 and CLOCK, which may contribute to the regulation of the sleep–wake cycle during epilepsy.H2020 Marie Skłodowska-Curie Actions, Grant/Award Number:766124 and 796600; Irish Research Council, Grant/Award Number:GOIPD/2020/806; Programa de Atracción de Talento de la Comunidad de Madrid, Grant/Award Number: 2020-T2/BMD- 20180; Science Foundation Ireland, Grant/Award Number:16/RC/3948 and 17/CDA/4708;Spanish Ministry of Economy and Competitiveness/Ministry of Science, Innovation and Universities, Grant/Award Number: PID2021- 123141OB- I00Peer reviewedJohn Wiley & SonsEuropean CommissionIrish Research CouncilComunidad de MadridScience Foundation IrelandMinisterio de Economía y Competitividad (España)Ministerio de Ciencia, Innovación y Universidades (España)Consejo Superior de Investigaciones Científicas [https://ror.org/02gfc7t72]2024202420232024info:eu-repo/semantics/articlehttp://purl.org/coar/resource_type/c_6501Publisher's versioninfo:eu-repo/semantics/publishedVersionhttp://hdl.handle.net/10261/346753reponame:DIGITAL.CSIC. Repositorio Institucional del CSICinstname:Consejo Superior de Investigaciones Científicas (CSIC)Inglés#PLACEHOLDER_PARENT_METADATA_VALUE##PLACEHOLDER_PARENT_METADATA_VALUE##PLACEHOLDER_PARENT_METADATA_VALUE##PLACEHOLDER_PARENT_METADATA_VALUE#info:eu-repo/grantAgreement/EC/H2020/766124info:eu-repo/grantAgreement/EC/H2020/796600info:eu-repo/grantAgreement/AEI/Plan Estatal de Investigación Científica y Técnica y de Innovación 2021-2023/PID2021-123141OB-I002020-T2/BMD-20180/ProgramadeAtraccióndeTalento-CMhttp://dx.doi.org/10.1111/epi.17736Síinfo:eu-repo/semantics/openAccessoai:digital.csic.es:10261/3467532026-05-22T06:33:51Z
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