Ataxia and epileptic seizures in mice lacking type 1 inositol 1,4,5-trisphosphate receptor

M. Matsumoto, T. Nakagawa, Takafumi Inoue, E. Nagata, K. Tanaka, H. Takano, O. Minowa, J. Kuno, Shinichi Sakakibara, M. Yamada, H. Yoneshima, A. Miyawaki, Y. Fukuuchi, T. Furuichi, H. Okano, K. Mikoshiba, T. Noda

Research output: Contribution to journalArticle

326 Citations (Scopus)

Abstract

The inositol 1,4,5-trisphosphate (InsP3) receptor acts as an InsP3-gated Ca2+ release channel in a variety of cell types. Type 1 InsP3 receptor (IP3R1) is the major neuronal member of the IP3R family in the central nervous system, predominantly enriched in cerebellar Purkinje cells but also concentrated in neurons in the hippocampal CA1 region, caudate-putamen, and cerebral cortex. Here we report that most IP3R1-deficient mice generated by gene targeting die in utero, and born animals have severe ataxia and tonic or tonic-clonic seizures and die by the weaning period. An electroencephalogram showed that they suffer from epilepsy, indicating that IP3R1 is essential for proper brain function. However, observation by light microscope of the haematoxylin-eosin staining of the brain and peripheral tissues of IP3R1-deficient mice showed no abnormality, and the unique electrophysiological properties of the cerebellar Purkinje cells of IP3R1 deficient mice were not severely impaired.

Original languageEnglish
Pages (from-to)168-171
Number of pages4
JournalNature
Volume379
Issue number6561
DOIs
Publication statusPublished - 1996 Jan 11
Externally publishedYes

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Inositol 1,4,5-Trisphosphate Receptors
Ataxia
Epilepsy
Purkinje Cells
Hippocampal CA1 Region
Gene Targeting
Putamen
Brain
Hematoxylin
Eosine Yellowish-(YS)
Weaning
Cerebral Cortex
Electroencephalography
Seizures
Central Nervous System
Observation
Staining and Labeling
Neurons
Light

ASJC Scopus subject areas

  • General

Cite this

Matsumoto, M., Nakagawa, T., Inoue, T., Nagata, E., Tanaka, K., Takano, H., ... Noda, T. (1996). Ataxia and epileptic seizures in mice lacking type 1 inositol 1,4,5-trisphosphate receptor. Nature, 379(6561), 168-171. https://doi.org/10.1038/379168a0

Ataxia and epileptic seizures in mice lacking type 1 inositol 1,4,5-trisphosphate receptor. / Matsumoto, M.; Nakagawa, T.; Inoue, Takafumi; Nagata, E.; Tanaka, K.; Takano, H.; Minowa, O.; Kuno, J.; Sakakibara, Shinichi; Yamada, M.; Yoneshima, H.; Miyawaki, A.; Fukuuchi, Y.; Furuichi, T.; Okano, H.; Mikoshiba, K.; Noda, T.

In: Nature, Vol. 379, No. 6561, 11.01.1996, p. 168-171.

Research output: Contribution to journalArticle

Matsumoto, M, Nakagawa, T, Inoue, T, Nagata, E, Tanaka, K, Takano, H, Minowa, O, Kuno, J, Sakakibara, S, Yamada, M, Yoneshima, H, Miyawaki, A, Fukuuchi, Y, Furuichi, T, Okano, H, Mikoshiba, K & Noda, T 1996, 'Ataxia and epileptic seizures in mice lacking type 1 inositol 1,4,5-trisphosphate receptor', Nature, vol. 379, no. 6561, pp. 168-171. https://doi.org/10.1038/379168a0
Matsumoto M, Nakagawa T, Inoue T, Nagata E, Tanaka K, Takano H et al. Ataxia and epileptic seizures in mice lacking type 1 inositol 1,4,5-trisphosphate receptor. Nature. 1996 Jan 11;379(6561):168-171. https://doi.org/10.1038/379168a0
Matsumoto, M. ; Nakagawa, T. ; Inoue, Takafumi ; Nagata, E. ; Tanaka, K. ; Takano, H. ; Minowa, O. ; Kuno, J. ; Sakakibara, Shinichi ; Yamada, M. ; Yoneshima, H. ; Miyawaki, A. ; Fukuuchi, Y. ; Furuichi, T. ; Okano, H. ; Mikoshiba, K. ; Noda, T. / Ataxia and epileptic seizures in mice lacking type 1 inositol 1,4,5-trisphosphate receptor. In: Nature. 1996 ; Vol. 379, No. 6561. pp. 168-171.
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