Zic2 controls the migration of specific neuronal populations in the developing forebrain

Human mutations in ZIC2 have been identified in patients with holoprosencephaly and schizophrenia. Similarly, Zic2 mutant mice exhibit holoprosencephaly in homozygosis and behavioral and morphological schizophrenic phenotypes associated with forebrain defects in heterozygosis. Despite the devastatin...

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Autores: Murillo, Blanca, Ruiz-Reig, Nuria, Herrera, Macarena, Herrera, Eloisa
Formato: artículo
Estado:Versión publicada
Fecha de publicación:2015
País:España
Recursos:Consejo Superior de Investigaciones Científicas (CSIC)
Repositorio:DIGITAL.CSIC. Repositorio Institucional del CSIC
OAI Identifier:oai:digital.csic.es:10261/338857
Acesso em linha:http://hdl.handle.net/10261/338857
Access Level:acceso abierto
Palavra-chave:Cajal–Retzius cells
Forebrain
LOT2 amygdaloid nucleus
Neuronal migration
vLGN
Zic2
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spelling Zic2 controls the migration of specific neuronal populations in the developing forebrainMurillo, BlancaRuiz-Reig, NuriaHerrera, MacarenaHerrera, EloisaCajal–Retzius cellsForebrainLOT2 amygdaloid nucleusNeuronal migrationvLGNZic2Human mutations in ZIC2 have been identified in patients with holoprosencephaly and schizophrenia. Similarly, Zic2 mutant mice exhibit holoprosencephaly in homozygosis and behavioral and morphological schizophrenic phenotypes associated with forebrain defects in heterozygosis. Despite the devastating effects of mutations in Zic2, the cellular and molecular mechanisms that provoke Zic2-deficiency phenotypes are yet unclear. Here, we report a novel role for this transcription factor in the migration of three different types of forebrain neurons: the Cajal-Retzius cells that populate the surface of the telencephalic vesicles, an amygdaloid group of cells originated in the caudal pole of the telencephalic pallium, and a cell population that travels from the prethalamic neuroepithelium to the ventral lateral geniculate nucleus. Our results also suggest that the receptor EphB1, previously identified as a Zic2 target, may mediate, at least partially, Zic2-dependent migratory events. According to these results, we propose that deficiencies in cell motility and guidance contribute to most of the forebrain pathologies associated with Zic2 mutations.[Significance statement]: Although the phenotype of Zic2 mutant individuals was reported more than 10 years ago, until now, the main function of this transcription factor during early development has not been precisely defined. Here, we reveal a previously unknown role for Zic2 in the migration of forebrain neurons such as Cajal-Retzius cells, interneurons moving to the ventral lateral geniculate nucleus, and neocortical cells going to the amygdala. We believe that the role of this transcription factor in certain populations of migratory cells contributes to defects in cortical layering and hypocellularity in the ventral LGN and amygdala and will contribute to our understanding of the devastating phenotypes associated with Zic2 mutations in both humans and mice.This work was supported by the Regional Government (Grant Prometeo 2012– 005 to E.H.), Spanish Ministry of Economy and Competitiveness (MINECO) (Grant BFU2010-16563 to E.H. and Grant BFU2010-17305 to A.F.), and the European Research Council (Grant ERC2011-StG201001109to E.H.). The Instituto de Neurociencias is a Severo Ochoa Excellence Center. B.M. and N.R.R. hold Formación de Personal Investigador (FPI) fellowships from the MINECO.Peer reviewedSociety for NeuroscienceMinisterio de Economía y Competitividad (España)Generalitat ValencianaEuropean CommissionEuropean Research CouncilConsejo Superior de Investigaciones Científicas [https://ror.org/02gfc7t72]202320232015info:eu-repo/semantics/articlehttp://purl.org/coar/resource_type/c_6501Publisher's versioninfo:eu-repo/semantics/publishedVersionapplication/pdfhttp://hdl.handle.net/10261/338857reponame:DIGITAL.CSIC. Repositorio Institucional del CSICinstname:Consejo Superior de Investigaciones Científicas (CSIC)Inglés#PLACEHOLDER_PARENT_METADATA_VALUE##PLACEHOLDER_PARENT_METADATA_VALUE#info:eu-repo/grantAgreement/MICINN//BFU2010-16563info:eu-repo/grantAgreement/MICINN//BFU2010-17305https://doi.org/10.1523/JNEUROSCI.0779-15.2015Síinfo:eu-repo/semantics/openAccessoai:digital.csic.es:10261/3388572026-05-22T06:33:51Z
dc.title.none.fl_str_mv Zic2 controls the migration of specific neuronal populations in the developing forebrain
title Zic2 controls the migration of specific neuronal populations in the developing forebrain
spellingShingle Zic2 controls the migration of specific neuronal populations in the developing forebrain
Murillo, Blanca
Cajal–Retzius cells
Forebrain
LOT2 amygdaloid nucleus
Neuronal migration
vLGN
Zic2
title_short Zic2 controls the migration of specific neuronal populations in the developing forebrain
title_full Zic2 controls the migration of specific neuronal populations in the developing forebrain
title_fullStr Zic2 controls the migration of specific neuronal populations in the developing forebrain
title_full_unstemmed Zic2 controls the migration of specific neuronal populations in the developing forebrain
title_sort Zic2 controls the migration of specific neuronal populations in the developing forebrain
dc.creator.none.fl_str_mv Murillo, Blanca
Ruiz-Reig, Nuria
Herrera, Macarena
Herrera, Eloisa
author Murillo, Blanca
author_facet Murillo, Blanca
Ruiz-Reig, Nuria
Herrera, Macarena
Herrera, Eloisa
author_role author
author2 Ruiz-Reig, Nuria
Herrera, Macarena
Herrera, Eloisa
author2_role author
author
author
dc.contributor.none.fl_str_mv Ministerio de Economía y Competitividad (España)
Generalitat Valenciana
European Commission
European Research Council
Consejo Superior de Investigaciones Científicas [https://ror.org/02gfc7t72]
dc.subject.none.fl_str_mv Cajal–Retzius cells
Forebrain
LOT2 amygdaloid nucleus
Neuronal migration
vLGN
Zic2
topic Cajal–Retzius cells
Forebrain
LOT2 amygdaloid nucleus
Neuronal migration
vLGN
Zic2
description Human mutations in ZIC2 have been identified in patients with holoprosencephaly and schizophrenia. Similarly, Zic2 mutant mice exhibit holoprosencephaly in homozygosis and behavioral and morphological schizophrenic phenotypes associated with forebrain defects in heterozygosis. Despite the devastating effects of mutations in Zic2, the cellular and molecular mechanisms that provoke Zic2-deficiency phenotypes are yet unclear. Here, we report a novel role for this transcription factor in the migration of three different types of forebrain neurons: the Cajal-Retzius cells that populate the surface of the telencephalic vesicles, an amygdaloid group of cells originated in the caudal pole of the telencephalic pallium, and a cell population that travels from the prethalamic neuroepithelium to the ventral lateral geniculate nucleus. Our results also suggest that the receptor EphB1, previously identified as a Zic2 target, may mediate, at least partially, Zic2-dependent migratory events. According to these results, we propose that deficiencies in cell motility and guidance contribute to most of the forebrain pathologies associated with Zic2 mutations.
publishDate 2015
dc.date.none.fl_str_mv 2015
2023
2023
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/10261/338857
url http://hdl.handle.net/10261/338857
dc.language.none.fl_str_mv Inglés
language_invalid_str_mv Inglés
dc.relation.none.fl_str_mv #PLACEHOLDER_PARENT_METADATA_VALUE#
#PLACEHOLDER_PARENT_METADATA_VALUE#
info:eu-repo/grantAgreement/MICINN//BFU2010-16563
info:eu-repo/grantAgreement/MICINN//BFU2010-17305
https://doi.org/10.1523/JNEUROSCI.0779-15.2015
Sí
dc.rights.none.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 Society for Neuroscience
publisher.none.fl_str_mv Society for Neuroscience
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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score 15.811543

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