G Protein-Coupled Receptor-Dependent Development of Human Frontal Cortex

Xianhua Piao; Sean S. Hill; Adria Bodell; Bernard S. Chang; Lina Basel-Vanagaite; Rachel Straussberg; William B. Dobyns; Bassam Qasrawi; Robin M. Winter; A. Micheil Innes; Thomas Voit; M. Elizabeth Ross; Jacques L. Michaud; Jean Claude Déscarie; A. James Barkovich; Christopher A. Walsh

doi:10.1126/science.1092780

G Protein-Coupled Receptor-Dependent Development of Human Frontal Cortex

Xianhua Piao, Sean S. Hill, Adria Bodell, Bernard S. Chang, Lina Basel-Vanagaite, Rachel Straussberg, William B. Dobyns, Bassam Qasrawi, Robin M. Winter, A. Micheil Innes, Thomas Voit, M. Elizabeth Ross, Jacques L. Michaud, Jean Claude Déscarie, A. James Barkovich, Christopher A. Walsh

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433 Scopus citations

Abstract

The mammalian cerebral cortex is characterized by complex patterns of anatomical and functional areas that differ markedly between species, but the molecular basis for this functional subdivision is largely unknown. Here, we show that mutations in GPR56, which encodes an orphan G protein-coupled receptor (GPCR) with a large extracellular domain, cause a human brain cortical malformation called bilateral frontoparietal polymicrogyria (BFPP). BFPP is characterized by disorganized cortical lamination that is most severe in frontal cortex. Our data suggest that GPCR signaling plays an essential role in regional development of human cerebral cortex.

Original language	English (US)
Pages (from-to)	2033-2036
Number of pages	4
Journal	Science
Volume	303
Issue number	5666
DOIs	https://doi.org/10.1126/science.1092780
State	Published - Mar 26 2004
Externally published	Yes

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Piao, X., Hill, S. S., Bodell, A., Chang, B. S., Basel-Vanagaite, L., Straussberg, R., Dobyns, W. B., Qasrawi, B., Winter, R. M., Innes, A. M., Voit, T., Ross, M. E., Michaud, J. L., Déscarie, J. C., Barkovich, A. J., & Walsh, C. A. (2004). G Protein-Coupled Receptor-Dependent Development of Human Frontal Cortex. Science, 303(5666), 2033-2036. https://doi.org/10.1126/science.1092780

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abstract = "The mammalian cerebral cortex is characterized by complex patterns of anatomical and functional areas that differ markedly between species, but the molecular basis for this functional subdivision is largely unknown. Here, we show that mutations in GPR56, which encodes an orphan G protein-coupled receptor (GPCR) with a large extracellular domain, cause a human brain cortical malformation called bilateral frontoparietal polymicrogyria (BFPP). BFPP is characterized by disorganized cortical lamination that is most severe in frontal cortex. Our data suggest that GPCR signaling plays an essential role in regional development of human cerebral cortex.",

author = "Xianhua Piao and Hill, {Sean S.} and Adria Bodell and Chang, {Bernard S.} and Lina Basel-Vanagaite and Rachel Straussberg and Dobyns, {William B.} and Bassam Qasrawi and Winter, {Robin M.} and Innes, {A. Micheil} and Thomas Voit and Ross, {M. Elizabeth} and Michaud, {Jacques L.} and D{\'e}scarie, {Jean Claude} and Barkovich, {A. James} and Walsh, {Christopher A.}",

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AU - Piao, Xianhua

AU - Hill, Sean S.

AU - Bodell, Adria

AU - Chang, Bernard S.

AU - Basel-Vanagaite, Lina

AU - Straussberg, Rachel

AU - Dobyns, William B.

AU - Qasrawi, Bassam

AU - Winter, Robin M.

AU - Innes, A. Micheil

AU - Voit, Thomas

AU - Ross, M. Elizabeth

AU - Michaud, Jacques L.

AU - Déscarie, Jean Claude

AU - Barkovich, A. James

AU - Walsh, Christopher A.

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AB - The mammalian cerebral cortex is characterized by complex patterns of anatomical and functional areas that differ markedly between species, but the molecular basis for this functional subdivision is largely unknown. Here, we show that mutations in GPR56, which encodes an orphan G protein-coupled receptor (GPCR) with a large extracellular domain, cause a human brain cortical malformation called bilateral frontoparietal polymicrogyria (BFPP). BFPP is characterized by disorganized cortical lamination that is most severe in frontal cortex. Our data suggest that GPCR signaling plays an essential role in regional development of human cerebral cortex.

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G Protein-Coupled Receptor-Dependent Development of Human Frontal Cortex

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