ZFIN ID: ZDB-PERS-030918-24
Hirata, Hiromi
Email: hihirata@chem.aoyama.ac.jp
Affiliation: Hirata Lab
Address: Professor Department of Chemistry and Biological Science Graduate School of Science and Engineering Aoyama Gakuin University 5-10-1 Fuchinobe, Sagamihara 252-5258, Japan
Country: Japan
Phone: +81-42-759-6235
Fax: +81-42-759-6386


Ogura, Y., Uehara, T., Ujibe, K., Yoshihashi, H., Yamada, M., Suzuki, H., Takenouchi, T., Kosaki, K., Hirata, H. (2022) The p.Thr395Met missense variant of NFIA found in a patient with intellectual disability is a defective variant. American journal of medical genetics. Part A. 188(4):1184-1192
Ujibe, K., Nishimura, K., Kashima, M., Hirata, H. (2021) Direct-TRI: High-throughput RNA-extracting Method for All Stages of Zebrafish Development. Bio-protocol. 11:e4136
Ogura, Y., Kaneko, R., Ujibe, K., Wakamatsu, Y., Hirata, H. (2021) Loss of αklotho causes reduced motor ability and short lifespan in zebrafish. Scientific Reports. 11:15090
Sadamitsu, K., Shigemitsu, L., Suzuki, M., Ito, D., Kashima, M., Hirata, H. (2021) Characterization of zebrafish GABAA receptor subunits. Scientific Reports. 11:6242
Wakamatsu, Y., Kashima, M., Hirata, H. (2020) A Reproducible Protocol to Measure the Critical Swimming Speed of Adult Zebrafish. Bio-protocol. 10:e3712
Ito, D., Kawazoe, Y., Sato, A., Uesugi, M., Hirata, H. (2020) Identification of the hypertension drug niflumic acid as a glycine receptor inhibitor. Scientific Reports. 10:13999
Maeta, M., Kataoka, M., Nishiya, Y., Ogino, K., Kashima, M., Hirata, H. (2020) RNA polymerase II subunit D is essential for zebrafish development. Scientific Reports. 10:13213
Rauschenberger, V., Wardenburg, N.V., Schaefer, N., Ogino, K., Hirata, H., Lillesaar, C., Kluck, C.J., Meinck, H.M., Borrmann, M., Weishaupt, A., Doppler, K., Wickel, J., Geis, C., Sommer, C., Villmann, C. (2020) GlyR autoantibodies impair receptor function and induce motor dysfunction. Annals of neurology. 88(3):544-561
Wakamatsu, Y., Ogino, K., Hirata, H. (2019) Swimming capability of zebrafish is governed by water temperature, caudal fin length and genetic background. Scientific Reports. 9:16307
Ogino, K., Yamada, K., Nishioka, T., Oda, Y., Kaibuchi, K., Hirata, H. (2019) Phosphorylation of gephyrin in zebrafish Mauthner cells governs glycine receptor clustering and behavioral desensitization to sound. The Journal of neuroscience : the official journal of the Society for Neuroscience. 39(45):8988-8997
Frints, S.G.M., Hennig, F., Colombo, R., Jacquemont, S., Terhal, P., Zimmerman, H.H., Hunt, D., Mendelsohn, B.A., Kordaß, U., Webster, R., Sinnema, M., Abdul-Rahman, O., Suckow, V., Deciphering Developmental Disorders (DDD) Study, Fernández-Jaén, A., van Roozendaal, K., Stevens, S.J.C., Macville, M.V.E., Al-Nasiry, S., van Gassen, K., Utzig, N., Koudijs, S.M., McGregor, L., Maas, S.M., Baralle, D., Dixit, A., Wieacker, P., Lee, M., Lee, A.S., Engle, E.C., Houge, G., Gradek, G.A., Douglas, A.G.L., Longman, C., Joss, S., Velasco, D., Hennekam, R.C., Hirata, H., Kalscheuer, V.M. (2019) Deleterious de novo variants of X-linked ZC4H2 in females cause a variable phenotype with neurogenic arthrogryposis multiplex congenita. Human Mutation. 40(12):2270-2285
Rho, S.S., Kobayashi, I., Oguri-Nakamura, E., Ando, K., Fujiwara, M., Kamimura, N., Hirata, H., Iida, A., Iwai, Y., Mochizuki, N., Fukuhara, S. (2019) Rap1b Promotes Notch-Signal-Mediated Hematopoietic Stem Cell Development by Enhancing Integrin-Mediated Cell Adhesion. Developmental Cell. 49(5):681-696.e6
Sugihara, M., Morito, D., Ainuki, S., Hirano, Y., Ogino, K., Kitamura, A., Hirata, H., Nagata, K. (2019) The AAA+ ATPase/ubiquitin ligase mysterin stabilizes cytoplasmic lipid droplets. The Journal of cell biology. 218:949-960
Low, S.E., Ito, D., Hirata, H. (2018) Characterization of the Zebrafish Glycine Receptor Family Reveals Insights Into Glycine Receptor Structure Function and Stoichiometry. Frontiers in molecular neuroscience. 11:286
Iida, A., Wang, Z., Hirata, H., Sehara-Fujisawa, A. (2018) Integrin β1 activity is required for cardiovascular formation in zebrafish. Genes to cells : devoted to molecular & cellular mechanisms. 23(11):938-951
Boubakri, M., Chaya, T., Hirata, H., Kajimura, N., Kuwahara, R., Ueno, A., Malicki, J., Furukawa, T., Omori, Y. (2016) Loss of ift122, a Retrograde IFT Complex Component, Leads to Slow, Progressive Photoreceptor Degeneration Due to Inefficient Opsin Transport. The Journal of biological chemistry. 291(47):24465-24474
Ogino, K., Hirata, H. (2016) Defects of the Glycinergic Synapse in Zebrafish. Frontiers in molecular neuroscience. 9:50
Knierim, E., Hirata, H., Wolf, N.I., Morales-Gonzalez, S., Schottmann, G., Tanaka, Y., Rudnik-Schöneborn, S., Orgeur, M., Zerres, K., Vogt, S., van Riesen, A., Gill, E., Seifert, F., Zwirner, A., Kirschner, J., Goebel, H.H., Hübner, C., Stricker, S., Meierhofer, D., Stenzel, W., Schuelke, M. (2016) Mutations in Subunits of the Activating Signal Cointegrator 1 Complex Are Associated with Prenatal Spinal Muscular Atrophy and Congenital Bone Fractures. American journal of human genetics. 98(3):473-89
Kotani, Y., Morito, D., Yamazaki, S., Ogino, K., Kawakami, K., Takashima, S., Hirata, H., Nagata, K. (2015) Neuromuscular regulation in zebrafish by a large AAA+ ATPase/ubiquitin ligase, mysterin/RNF213. Scientific Reports. 5:16161
Stödberg, T., McTague, A., Ruiz, A.J., Hirata, H., Zhen, J., Long, P., Farabella, I., Meyer, E., Kawahara, A., Vassallo, G., Stivaros, S.M., Bjursell, M.K., Stranneheim, H., Tigerschiöld, S., Persson, B., Bangash, I., Das, K., Hughes, D., Lesko, N., Lundeberg, J., Scott, R.C., Poduri, A., Scheffer, I.E., Smith, H., Gissen, P., Schorge, S., Reith, M.E., Topf, M., Kullmann, D.M., Harvey, R.J., Wedell, A., Kurian, M.A. (2015) Mutations in SLC12A5 in epilepsy of infancy with migrating focal seizures. Nature communications. 6:8038
Ogino, K., Low, S.E., Yamada, K., Saint-Amant, L., Zhou, W., Muto, A., Asakawa, K., Nakai, J., Kawakami, K., Kuwada, J.Y., Hirata, H. (2015) RING finger protein 121 facilitates the degradation and membrane localization of voltage-gated sodium channels. Proceedings of the National Academy of Sciences of the United States of America. 112(9):2859-64
Watanabe, T., Shimazaki, T., Mishiro, A., Suzuki, T., Hirata, H., Tanimoto, M., and Oda, Y. (2014) Coexpression of auxiliary Kvbeta2 subunits with Kv1.1 channels is required for developmental acquisition of unique firing properties of zebrafish Mauthner cells. Journal of neurophysiology. 111(6):1153-64
Hirata, H., Ogino, K., Yamada, K., Leacock, S., and Harvey, R.J. (2013) Defective Escape Behavior in DEAH-Box RNA Helicase Mutants Improved by Restoring Glycine Receptor Expression. The Journal of neuroscience : the official journal of the Society for Neuroscience. 33(37):14638-14644
Satou, C., Kimura, Y., Hirata, H., Suster, M.L., Kawakami, K., and Higashijima, S. (2013) Transgenic tools to characterize neuronal properties of discrete populations of zebrafish neurons. Development (Cambridge, England). 140(18):3927-3931
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., Hübner, C., Ropers, H.H., Gecz, J., Schuelke, M., Laumonnier, F., and Kalscheuer, V.M. (2013) ZC4H2 Mutations Are Associated with Arthrogryposis Multiplex Congenita and Intellectual Disability through Impairment of Central and Peripheral Synaptic Plasticity. American journal of human genetics. 92(5):681-695
Yamanaka, I., Miki, M., Asakawa, K., Kawakami, K., Oda, Y., and Hirata, H. (2013) Glycinergic transmission and postsynaptic activation of CaMKII are required for glycine receptor clustering in vivo. Genes to cells : devoted to molecular & cellular mechanisms. 18(3):211-224
Horstick, E.J., Linsley, J.W., Dowling, J.J., Hauser, M.A., McDonald, K.K., Ashley-Koch, A., Saint-Amant, L., Satish, A., Cui, W.W., Zhou, W., Sprague, S.M., Stamm, D.S., Powell, C.M., Speer, M.C., Franzini-Armstrong, C., Hirata, H., and Kuwada, J.Y. (2013) Stac3 is a component of the excitation-contraction coupling machinery and mutated in Native American myopathy. Nature communications. 4:1952
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. (2012) Connexin39.9 is necessary for coordinated activation of slow-twitch muscle and normal behavior in zebrafish. The Journal of biological chemistry. 287(2):1080-9
Hirata, H., Takahashi, M., Yamada, K., and Ogino, K. (2011) The biological role of the glycinergic synapse in early zebrafish motility. Neuroscience research. 71(1):1-11
Low, S.E., Amburgey, K., Horstick, E., Linsley, J., Sprague, S.M., Cui, W.W., Zhou, W., Hirata, H., Saint-Amant, L., Hume, R.I., and Kuwada, J.Y. (2011) TRPM7 Is Required within Zebrafish Sensory Neurons for the Activation of Touch-Evoked Escape Behaviors. The Journal of neuroscience : the official journal of the Society for Neuroscience. 31(32):11633-11644
Naganawa, Y., and Hirata, H. (2011) Developmental transition of touch response from slow muscle-mediated coilings to fast muscle-mediated burst swimming in zebrafish. Developmental Biology. 355(2):194-204
Ogino, K., Ramsden, S.L., Keib, N., Schwarz, G., Harvey, R.J., and Hirata, H. (2011) Duplicated Gephyrin Genes Showing Distinct Tissue Distribution and Alternative Splicing Patterns Mediate Molybdenum Cofactor Biosynthesis, Glycine Receptor Clustering, and Escape Behavior in Zebrafish. The Journal of biological chemistry. 286(1):806-817
Low, S.E., Ryan, J., Sprague, S.M., Hirata, H., Cui, W.W., Zhou, W., Hume, R.I., Kuwada, J.Y., and Saint-Amant, L. (2010) touche Is Required for Touch-Evoked Generator Potentials within Vertebrate Sensory Neurons. The Journal of neuroscience : the official journal of the Society for Neuroscience. 30(28):9359-9367
Low, S.E., Zhou, W., Choong, X., Saint-Amant, L., Sprague, S.M., Hirata, H., Cui, W.W., Hume, R.I., and Kuwada, J.Y. (2010) Na(V)1.6a is required for normal activation of motor circuits normally excited by tactile stimulation. Developmental Neurobiology. 70(7):508-522
Nakano, Y., Fujita, M., Ogino, K., Saint-Amant, L., Kinoshita, T., Oda, Y., and Hirata, H. (2010) Biogenesis of GPI-anchored proteins is essential for surface expression of sodium channels in zebrafish Rohon-Beard neurons to respond to mechanosensory stimulation. Development (Cambridge, England). 137(10):1689-1698
Hirata, H., Carta, E., Yamanaka, I., Harvey, R.J., and Kuwada, J.Y. (2010) Defective glycinergic synaptic transmission in zebrafish motility mutants. Frontiers in molecular neuroscience. 2:26
Zhou, W., Horstick, E.J., Hirata, H., and Kuwada, J.Y. (2008) Identification and expression of voltage-gated calcium channel beta subunits in Zebrafish. Developmental dynamics : an official publication of the American Association of Anatomists. 237(12):3842-3852
Saint-Amant, L., Sprague, S.M., Hirata, H., Li, Q., Cui, W.W., Zhou, W., Poudou, O., Hume, R.I., and Kuwada, J.Y. (2008) The zebrafish ennui behavioral mutation disrupts acetylcholine receptor localization and motor axon stability. Developmental Neurobiology. 68(1):45-61
Hirata, H., Watanabe, T., Hatakeyama, J., Sprague, S.M., Saint-Amant, L., Nagashima, A., Cui, W.W., Zhou, W., and Kuwada, J.Y. (2007) Zebrafish relatively relaxed mutants have a ryanodine receptor defect, show slow swimming and provide a model of multi-minicore disease. Development (Cambridge, England). 134(15):2771-2781
Zhou, W., Saint-Amant, L., Hirata, H., Cui, W.W., Sprague, S.M., and Kuwada, J.Y. (2006) Non-sense mutations in the dihydropyridine receptor beta1 gene, CACNB1, paralyze zebrafish relaxed mutants. Cell Calcium. 39(3):227-236
Cui, W.W., Low, S.E., Hirata, H., Saint-Amant, L., Geisler, R., Hume, R.I., and Kuwada, J.Y. (2005) The zebrafish shocked gene encodes a glycine transporter and is essential for the function of early neural circuits in the CNS. The Journal of neuroscience : the official journal of the Society for Neuroscience. 25(28):6610-6620
Hirata, H., Saint-Amant, L., Downes, G.B., Cui, W.W., Zhou, W., Granato, M., and Kuwada, J.Y. (2005) Zebrafish bandoneon mutants display behavioral defects due to a mutation in the glycine receptor β-subunit. Proceedings of the National Academy of Sciences of the United States of America. 102(23):8345-8350
Hirata, H., Saint-Amant, L., Waterbury, J., Cui, W., Zhou, W., Li, Q., Goldman, D., Granato, M., and Kuwada, J.Y. (2004) accordion, a zebrafish behavioral mutant, has a muscle relaxation defect due to a mutation in the ATPase Ca2+ pump SERCA1. Development (Cambridge, England). 131(21):5457-5468