GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development

Jeong, Sung-Jin; Luo, Rong; Singer, Kathleen; Giera, Stefanie; Kreidberg, Jordan; Kiyozumi, Daiji; Shimono, Chisei; Sekiguchi, Kiyotoshi; Piao, Xianhua

doi:https://doi.org/10.1371/journal.pone.0068781

GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development

Jeong, Sung-Jin; Luo, Rong; Singer, Kathleen et al.

PLOS ONE, 2013, 8(7), e68781

Number of Access:663（2025-08-18 12:35 Counts）

Identifier to cite or link to this item: https://hdl.handle.net/11094/71798

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Title	GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development
Creator	CreatorName Jeong, Sung-Jin
	CreatorName Luo, Rong
	CreatorName Singer, Kathleen
	CreatorName Giera, Stefanie
	CreatorName Kreidberg, Jordan
	NRID 1000070452430 1000070452430 ORCID 0000-0003-3289-227X 0000-0003-3289-227X researchmap permalink dkiyozumi dkiyozumi Scopus Author ID 6506341053 6506341053 CreatorName Kiyozumi, Daiji
	CreatorName Shimono, Chisei
	NRID 1000050187845 1000050187845 researchmap permalink read0054833 read0054833 Scopus Author ID 7201662709 7201662709 ORCID 0000-0002-6433-1410 0000-0002-6433-1410 CreatorName Sekiguchi, Kiyotoshi
	CreatorName Piao, Xianhua
Description	Jeong S-J, Luo R, Singer K, Giera S, Kreidberg J, Kiyozumi D, et al. (2013) GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development. PLoS ONE 8(7): e68781. https://doi.org/10.1371/journal.pone.0068781
Abstract	Loss of function mutations in GPR56, which encodes a G protein-coupled receptor, cause a specific human brain malformation called bilateral frontoparietal polymicrogyria (BFPP). Studies from BFPP postmortem brain tissue and Gpr56 knockout mice have previously showed that GPR56 deletion leads to breaches in the pial basement membrane (BM) and neuronal ectopias during cerebral cortical development. Since α3β1 integrin also plays a role in pial BM assembly and maintenance, we evaluated whether it functions together with GPR56 in regulating the same developmental process. We reveal that loss of α3 integrin enhances the cortical phenotype associated with Gpr56 deletion, and that neuronal overmigration through a breached pial BM occurs earlier in double knockout than in Gpr56 single knockout mice. These observations provide compelling evidence of the synergism of GPR56 and α3β1 integrin in regulating the development of cerebral cortex.
Publisher	PLOS
Source Title	PLOS ONE
Volume (Issue)	8(7)
Page	e68781
Date of Issued	2013-07
Language	English
Handle URL	https://hdl.handle.net/11094/71798
eISSN	19326203
Relation.isIdenticalTo	DOI (Publisher Version) https://doi.org/10.1371/journal.pone.0068781
Access Rights	open access
CopyRight	© 2013 Jeong et al. This article is licensed under a Creative Commons Attribution 4.0 International License.
oaire:version	Version of Record
Category	学術雑誌論文 Journal Article

Resource Type	journal article
Local Type	Journal Article
DCMI Type	Text
DCTERMS.bibliographicCitation	PLOS ONE. 2013, 8(7), p. e68781
DC.title	GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development
DC.creator	Jeong, Sung-Jin
	Luo, Rong
	Singer, Kathleen
	Giera, Stefanie
	Kreidberg, Jordan
	Kiyozumi, Daiji
	Shimono, Chisei
	Sekiguchi, Kiyotoshi
	Piao, Xianhua
DC.publisher	PLOS
DC.language' scheme='DCTERMS.RFC1766	English
DC.type' scheme='DCTERMS.DCMIType	Text
DCTERMS.issued' scheme='DCTERMS.W3CDTF	2013-07
DC.identifier	https://doi.org/10.1371/journal.pone.0068781
DC.description	Jeong S-J, Luo R, Singer K, Giera S, Kreidberg J, Kiyozumi D, et al. (2013) GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development. PLoS ONE 8(7): e68781. https://doi.org/10.1371/journal.pone.0068781
DCTERMS.abstract	Loss of function mutations in GPR56, which encodes a G protein-coupled receptor, cause a specific human brain malformation called bilateral frontoparietal polymicrogyria (BFPP). Studies from BFPP postmortem brain tissue and Gpr56 knockout mice have previously showed that GPR56 deletion leads to breaches in the pial basement membrane (BM) and neuronal ectopias during cerebral cortical development. Since α3β1 integrin also plays a role in pial BM assembly and maintenance, we evaluated whether it functions together with GPR56 in regulating the same developmental process. We reveal that loss of α3 integrin enhances the cortical phenotype associated with Gpr56 deletion, and that neuronal overmigration through a breached pial BM occurs earlier in double knockout than in Gpr56 single knockout mice. These observations provide compelling evidence of the synergism of GPR56 and α3β1 integrin in regulating the development of cerebral cortex.
DC.rights	© 2013 Jeong et al. This article is licensed under a Creative Commons Attribution 4.0 International License.
DC.format	71798
citation_title	GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development
citation_author	Jeong, Sung-Jin
	Luo, Rong
	Singer, Kathleen
	Giera, Stefanie
	Kreidberg, Jordan
	Kiyozumi, Daiji
	Shimono, Chisei
	Sekiguchi, Kiyotoshi
	Piao, Xianhua
citation_publisher	PLOS
citation_language	English
citation_date	2013-07
citation_journal_title	PLOS ONE
citation_volume	8
citation_issue	7
citation_firstpage	e68781
citation_doi	https://doi.org/10.1371/journal.pone.0068781
citation_public_url	https://hdl.handle.net/11094/71798

GPR56 Functions Together with α3β1 Integrin in Regulating Cerebral Cortical Development

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