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dc.contributor.authorDamián, Juan Pablo
dc.contributor.authorVázquez Alberdi, Lucia
dc.contributor.authorCanclini, Lucía
dc.contributor.authorRosso, Gonzalo
dc.contributor.authorBravo, Silvia Olivera
dc.contributor.authorMartínez, Mariana
dc.contributor.authorUriarte, Natalia
dc.contributor.authorRuiz, Paul
dc.contributor.authorCalero, Miguel 
dc.contributor.authorDi Tomaso, María Vittoria
dc.contributor.authorKun, Alejandra
dc.date.accessioned2021-05-13T20:05:15Z
dc.date.available2021-05-13T20:05:15Z
dc.date.issued2021-04-19
dc.identifier.citationBiomolecules. 2021 Apr 19;11(4):601.es_ES
dc.identifier.urihttp://hdl.handle.net/20.500.12105/12959
dc.description.abstractCharcot-Marie-Tooth (CMT) type 1 disease is the most common human hereditary demyelinating neuropathy. Mutations in pmp22 cause about 70% of all CMT1. Trembler-J (TrJ/+) mice are an animal model of CMT1E, having the same spontaneous pmp22 mutation that is found in humans. We compared the behavior profile of TrJ/+ and +/+ (wild-type) in open-field and elevated-plus-maze anxiety tests. In these tests, TrJ/+ showed an exclusive head shake movement, a lower frequency of rearing, but a greater frequency of grooming. In elevated-plus-maze, TrJ/+ defecate more frequently, performed fewer total entries, and have fewer entries to closed arms. These hippocampus-associated behaviors in TrJ/+ are consistent with increased anxiety levels. The expression of pmp22 and soluble PMP22 were evaluated in E17-hippocampal neurons and adult hippocampus by in situ hybridization and successive immunohistochemistry. Likewise, the expression of pmp22 was confirmed by RT-qPCR in the entire isolated hippocampi of both genotypes. Moreover, the presence of aggregated PMP22 was evidenced in unmasked granular hippocampal adult neurons and shows genotypic differences. We showed for the first time a behavior profile trait associated with anxiety and a differential expression of pmp22/PMP22 in hippocampal neurons of TrJ/+ and +/+ mice, demonstrating the involvement at the central level in an animal model of peripheral neuropathy (CMT1E).es_ES
dc.description.sponsorshipThis research was funded by the Comisión Sectorial de Investigación Científica de la Universidad de la República; Agencia Nacional de Investigación e Innovación (ANII) and the Programa de Desarrollo de Ciencias Básicas (PEDECIBA).es_ES
dc.language.isoenges_ES
dc.publisherMultidisciplinary Digital Publishing Institute (MDPI) es_ES
dc.relation.isversionofPublisher's versiones_ES
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/*
dc.subjectCA3 neuronses_ES
dc.subjectCharcot–Marie–Toothes_ES
dc.subjectTrembler-Jes_ES
dc.subjectAnxietyes_ES
dc.subjectHippocampuses_ES
dc.subjectPeripheral-myelin-protein-22es_ES
dc.titleCentral Alteration in Peripheral Neuropathy of Trembler-J Mice: Hippocampal pmp22 Expression and Behavioral Profile in Anxiety Testses_ES
dc.typeArtículoes_ES
dc.rights.licenseAtribución 4.0 Internacional*
dc.identifier.pubmedID33921657es_ES
dc.format.volume11es_ES
dc.format.number4es_ES
dc.format.page601es_ES
dc.identifier.doi10.3390/biom11040601es_ES
dc.contributor.funderUniversity of the Republic (Uruguay)
dc.contributor.funderAgencia Nacional de Investigación e Innovación (Uruguay)
dc.contributor.funderPrograma de Desarrollo de las Ciencias Básicas (Uruguay)
dc.description.peerreviewedes_ES
dc.identifier.e-issn2218-273Xes_ES
dc.relation.publisherversionhttps://doi.org/10.3390/biom11040601es_ES
dc.identifier.journalBiomoleculeses_ES
dc.repisalud.centroISCIII::Unidad Funcional de Investigación de Enfermedades Crónicas (UFIEC)es_ES
dc.repisalud.institucionISCIIIes_ES
dc.rights.accessRightsinfo:eu-repo/semantics/openAccesses_ES


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Atribución 4.0 Internacional
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