Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family

Taisuke Miyazaki; Kouichi Hashimoto; Atsushi Uda; Hiroyuki Sakagami; Yoshitaka Nakamura; Shin ya Saito; Miyuki Nishi; Hideaki Kume; Akira Tohgo; Izumi Kaneko; Hisatake Kondo; Kohji Fukunaga; Masanobu Kano; Masahiko Watanabe; Hiroshi Takeshima

doi:10.1016/j.febslet.2006.06.043

Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family

Taisuke Miyazaki, Kouichi Hashimoto, Atsushi Uda, Hiroyuki Sakagami, Yoshitaka Nakamura, Shin ya Saito, Miyuki Nishi, Hideaki Kume, Akira Tohgo, Izumi Kaneko, Hisatake Kondo, Kohji Fukunaga, Masanobu Kano, Masahiko Watanabe, Hiroshi Takeshima

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57 Citations (Scopus)

Abstract

By DNA cloning, we have identified the BSRP (brain-specific receptor-like proteins) family of three members in mammalian genomes. BSRPs were predominantly expressed in the soma and dendrites of neurons and localized in the endoplasmic reticulum (ER). Expression levels of BSRPs seemed to fluctuate greatly during postnatal cerebellar maturation. Triple-knockout mice lacking BSRP members exhibited motor discoordination, and Purkinje cells (PCs) were often innervated by multiple climbing fibers with different neuronal origins in the mutant cerebellum. Moreover, the phosphorylation levels of protein kinase Cα (PKCα) were significantly downregulated in the mutant cerebellum. Because cerebellar maturation and plasticity require metabotropic glutamate receptor signaling and resulting PKC activation, BSRPs are likely involved in ER functions supporting PKCα activation in PCs.

Original language	English
Pages (from-to)	4057-4064
Number of pages	8
Journal	FEBS Letters
Volume	580
Issue number	17
DOIs	https://doi.org/10.1016/j.febslet.2006.06.043
Publication status	Published - 2006 Jul 24
Externally published	Yes

Keywords

Cerebellum
Endoplasmic reticulum
Knockout mouse
Protein kinase C

ASJC Scopus subject areas

Biophysics
Structural Biology
Biochemistry
Molecular Biology
Genetics
Cell Biology

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10.1016/j.febslet.2006.06.043

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Miyazaki, T., Hashimoto, K., Uda, A., Sakagami, H., Nakamura, Y., Saito, S. Y., Nishi, M., Kume, H., Tohgo, A., Kaneko, I., Kondo, H., Fukunaga, K., Kano, M., Watanabe, M., & Takeshima, H. (2006). Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family. FEBS Letters, 580(17), 4057-4064. https://doi.org/10.1016/j.febslet.2006.06.043

Miyazaki, T, Hashimoto, K, Uda, A, Sakagami, H, Nakamura, Y, Saito, SY, Nishi, M, Kume, H, Tohgo, A, Kaneko, I, Kondo, H, Fukunaga, K, Kano, M, Watanabe, M & Takeshima, H 2006, 'Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family', FEBS Letters, vol. 580, no. 17, pp. 4057-4064. https://doi.org/10.1016/j.febslet.2006.06.043

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title = "Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family",

abstract = "By DNA cloning, we have identified the BSRP (brain-specific receptor-like proteins) family of three members in mammalian genomes. BSRPs were predominantly expressed in the soma and dendrites of neurons and localized in the endoplasmic reticulum (ER). Expression levels of BSRPs seemed to fluctuate greatly during postnatal cerebellar maturation. Triple-knockout mice lacking BSRP members exhibited motor discoordination, and Purkinje cells (PCs) were often innervated by multiple climbing fibers with different neuronal origins in the mutant cerebellum. Moreover, the phosphorylation levels of protein kinase Cα (PKCα) were significantly downregulated in the mutant cerebellum. Because cerebellar maturation and plasticity require metabotropic glutamate receptor signaling and resulting PKC activation, BSRPs are likely involved in ER functions supporting PKCα activation in PCs.",

keywords = "Cerebellum, Endoplasmic reticulum, Knockout mouse, Protein kinase C",

author = "Taisuke Miyazaki and Kouichi Hashimoto and Atsushi Uda and Hiroyuki Sakagami and Yoshitaka Nakamura and Saito, {Shin ya} and Miyuki Nishi and Hideaki Kume and Akira Tohgo and Izumi Kaneko and Hisatake Kondo and Kohji Fukunaga and Masanobu Kano and Masahiko Watanabe and Hiroshi Takeshima",

note = "Funding Information: We thank Miyuki Kameyama for technical assistance in mutant mouse generation, Hidemi Shimizu for production of anti-GFAP antibody, and Motokazu Uchigashima for immunoblot analysis. This work was supported in part by grants from the Ministry of Education, Culture, Sports, Science and Technology of Japan, the Ministry of Health and Welfare of Japan, and the Mitsubishi Foundation. ",

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doi = "10.1016/j.febslet.2006.06.043",

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T1 - Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family

AU - Miyazaki, Taisuke

AU - Hashimoto, Kouichi

AU - Uda, Atsushi

AU - Sakagami, Hiroyuki

AU - Nakamura, Yoshitaka

AU - Saito, Shin ya

AU - Nishi, Miyuki

AU - Kume, Hideaki

AU - Tohgo, Akira

AU - Kaneko, Izumi

AU - Kondo, Hisatake

AU - Fukunaga, Kohji

AU - Kano, Masanobu

AU - Watanabe, Masahiko

AU - Takeshima, Hiroshi

N1 - Funding Information: We thank Miyuki Kameyama for technical assistance in mutant mouse generation, Hidemi Shimizu for production of anti-GFAP antibody, and Motokazu Uchigashima for immunoblot analysis. This work was supported in part by grants from the Ministry of Education, Culture, Sports, Science and Technology of Japan, the Ministry of Health and Welfare of Japan, and the Mitsubishi Foundation.

PY - 2006/7/24

Y1 - 2006/7/24

N2 - By DNA cloning, we have identified the BSRP (brain-specific receptor-like proteins) family of three members in mammalian genomes. BSRPs were predominantly expressed in the soma and dendrites of neurons and localized in the endoplasmic reticulum (ER). Expression levels of BSRPs seemed to fluctuate greatly during postnatal cerebellar maturation. Triple-knockout mice lacking BSRP members exhibited motor discoordination, and Purkinje cells (PCs) were often innervated by multiple climbing fibers with different neuronal origins in the mutant cerebellum. Moreover, the phosphorylation levels of protein kinase Cα (PKCα) were significantly downregulated in the mutant cerebellum. Because cerebellar maturation and plasticity require metabotropic glutamate receptor signaling and resulting PKC activation, BSRPs are likely involved in ER functions supporting PKCα activation in PCs.

AB - By DNA cloning, we have identified the BSRP (brain-specific receptor-like proteins) family of three members in mammalian genomes. BSRPs were predominantly expressed in the soma and dendrites of neurons and localized in the endoplasmic reticulum (ER). Expression levels of BSRPs seemed to fluctuate greatly during postnatal cerebellar maturation. Triple-knockout mice lacking BSRP members exhibited motor discoordination, and Purkinje cells (PCs) were often innervated by multiple climbing fibers with different neuronal origins in the mutant cerebellum. Moreover, the phosphorylation levels of protein kinase Cα (PKCα) were significantly downregulated in the mutant cerebellum. Because cerebellar maturation and plasticity require metabotropic glutamate receptor signaling and resulting PKC activation, BSRPs are likely involved in ER functions supporting PKCα activation in PCs.

KW - Cerebellum

KW - Endoplasmic reticulum

KW - Knockout mouse

KW - Protein kinase C

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Disturbance of cerebellar synaptic maturation in mutant mice lacking BSRPs, a novel brain-specific receptor-like protein family

Abstract

Keywords

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