The adhesion G protein-coupled receptor GPR56 is a cell-autonomous regulator of oligodendrocyte development

Stefanie Giera, Yiyu Deng, Rong Luo, Sarah D. Ackerman, Amit Mogha, Kelly R. Monk, Yanqin Ying, Sung-Jin Jeong, Manabu Makinodan, Allison R. Bialas, Bernard S. Chang, Beth Stevens, Gabriel Corfas and Xianhua Piao ()
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Stefanie Giera: Boston Children’s Hospital and Harvard Medical School
Yiyu Deng: Boston Children’s Hospital and Harvard Medical School
Rong Luo: Boston Children’s Hospital and Harvard Medical School
Sarah D. Ackerman: Washington University School of Medicine
Amit Mogha: Washington University School of Medicine
Kelly R. Monk: Washington University School of Medicine
Yanqin Ying: Boston Children’s Hospital and Harvard Medical School
Sung-Jin Jeong: Boston Children’s Hospital and Harvard Medical School
Manabu Makinodan: F.M. Kirby Neurobiology Center, Children’s Hospital, Harvard Medical School
Allison R. Bialas: F.M. Kirby Neurobiology Center, Children’s Hospital, Harvard Medical School
Bernard S. Chang: Comprehensive Epilepsy Center, Beth Israel Deaconess Medical Center and Harvard Medical School
Beth Stevens: F.M. Kirby Neurobiology Center, Children’s Hospital, Harvard Medical School
Gabriel Corfas: F.M. Kirby Neurobiology Center, Children’s Hospital, Harvard Medical School
Xianhua Piao: Boston Children’s Hospital and Harvard Medical School

Nature Communications, 2015, vol. 6, issue 1, 1-12

Abstract: Abstract Mutations in GPR56, a member of the adhesion G protein-coupled receptor family, cause a human brain malformation called bilateral frontoparietal polymicrogyria (BFPP). Magnetic resonance imaging (MRI) of BFPP brains reveals myelination defects in addition to brain malformation. However, the cellular role of GPR56 in oligodendrocyte development remains unknown. Here, we demonstrate that loss of Gpr56 leads to hypomyelination of the central nervous system in mice. GPR56 levels are abundant throughout early stages of oligodendrocyte development, but are downregulated in myelinating oligodendrocytes. Gpr56-knockout mice manifest with decreased oligodendrocyte precursor cell (OPC) proliferation and diminished levels of active RhoA, leading to fewer mature oligodendrocytes and a reduced number of myelinated axons in the corpus callosum and optic nerves. Conditional ablation of Gpr56 in OPCs leads to a reduced number of mature oligodendrocytes as seen in constitutive knockout of Gpr56. Together, our data define GPR56 as a cell-autonomous regulator of oligodendrocyte development.

Date: 2015
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DOI: 10.1038/ncomms7121

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