Physiological genetics of primary sensory neurons

2012 Fiscal Year Final Research Report

• Project/Area Number: 23700477
• Research Category: Grant-in-Aid for Young Scientists (B)
• Allocation Type: Multi-year Fund
• Research Field: Neurophysiology and muscle physiology
• Research Institution: National Institute of Genetics
• Principal Investigator: HIRATA Hiromi 国立遺伝学研究所, 新分野創造センター, 准教授 (60402450)
• Project Period (FY): 2011 – 2012
• Keywords: 体性感覚

Research Abstract

We identified an E3 ubiquitin ligase ring finger protein 121 as an essential gene for mechanoreception. We also revealed that this gene is essential for voltage-gated sodium channel function. Zebrafish embryos that lack the ring finger protein 121 did not respond to either tactile stimulus or mustard oil, thereby demonstrating that the ring finger protein 121 is indispensable for mechanoreception and chemical reception, which are essential for survival of animals.

Research Products

• [Journal Article] Mutations of ZC4H2 are associated with arthrogryposis multiplex congenita and intellectual disability and through impairment of central and peripheral synaptic plasticity (2013)
  • Author(s): Hirata, H., Nanda, I., van Riesen, A., McMichael, G., Hu, H., Hambrock, M., Papon, M.-A., Fischer, U., Marouillat, S., Ding, C., Alirol, S., Bienek, M., Preisler-Adams, S., Grimme, A., Seelow, D., Webster, R., Haan, E., MacLennan, A., Stenzel, W., Yap, T. Y., Gardner, A., Nguyen, L. S., Shaw, M., Lebrun, N., Haas, S. A., Kress, W., Haaf, T., Schellenberger, E., Chelly, J., Viot, G., Shaffer, L. G., Rosenfeld, J. A., Kramer, N., Falk, R., El-Khechen, D., Escobar, L. F., Hennekam, R., Wieacker, P., Hubner, C., Ropers, H.-H., Gecz, J., Schuelke, M., Laumonnier, F. and Kalscheuer, V. M
  • Journal Title: Am. J. Hum. Genet Volume: 92 Pages: 681-695
  • DOI: doi:10.1016/j.ajhg.2013.03.021
  • Peer Reviewed
• [Journal Article] Glycinergic transmission and postsynaptic activation of CaMKII are required for glycine receptor clustering in vivo (2013)
  • Author(s): Yamanaka, I., Miki, M., Asakawa, K., Kawakami, K., Oda, Y. and Hirata, H
  • Journal Title: Genes Cells Volume: 18 Pages: 211-224
  • DOI: doi:10.1111/gtc.12032
  • Peer Reviewed
• [Journal Article] Connexin39.9 is necessary for coordinated activation of slow-twitch muscle and normal behavior in zebrafish (2012)
  • Author(s): Hirata, H., Wen, H., Kawakami, Y., Naganawa, Y., Ogino, K., Yamada, K., Saint-Amant, L., Low, S. E., Cui, W. W., Zhou, W., Sprague, S. M., Asakawa, K., Muto, A., Kawakami, K. and Kuwada, J. Y
  • Journal Title: J. Biol. Chem Volume: 287 Pages: 1080-1089
  • DOI: doi:10.1074/jbc.M111.308205
  • Peer Reviewed
• [Presentation] Formation and function of glycinergic synapse in zebrafish (2013)
  • Author(s): Hirata, H
  • Organizer: International Institute for Advanced Studies Research Project "Novel developments on the study of life and biological systems based on genome engineering and imaging science" The First Symposium
  • Place of Presentation: Kizugawa
  • Year and Date: 2013-02-23
• [Presentation] Defective escape behavior in DEAH-box RNA helicase Dhx37 mutant zebrafish improved by restoring glycine receptor expression (2013)
  • Author(s): Hirata, H., Ogino, K., Yamada, K., and Harvey, R. J
  • Organizer: The 5th Strategic Conference of Zebrafish Investigators
  • Place of Presentation: California, USA
  • Year and Date: 2013-01-21
• [Presentation] Slow-twitch and fast-twitch muscle defects in zebrafish (2012)
  • Author(s): Hirata, H
  • Organizer: Cold Spring Harbor Asia Conference
  • Place of Presentation: China
  • Year and Date: 2012-04-19
• [Book] グリシン受容体の構造と機能 (2012)
  • Author(s): 平田普三
  • Publisher: Clinical Neuroscience