In 2011-2012, we published eight papers. Among them, two were focused on the neuromuscular junctions. In Mongeon et al. published in the Journal of General Physiology, we revealed the differential composition of nicotinic ACh receptors (AChR) in two kinds of skeletal muscles, slow muscle and fast muscle. In Park et al. published in the Journal of Neuroscience, we discovered that AChRs, which are classically considered a passive player in the synapse formation, plays an active role, by allowing the exit of raspy molecule from the Golgi complex. In Papke at al. published in Biochemical Pharmacology, we examined the function of nicotinic AChRs in zebrafish, including muscle type and neural type. We compared the pharmacological profile of zebrafish receptors to those of mammalian receptors. Three papers were published resulting from the forward genetics screening performed in the lab. A gene trap line with an insertion of Red Fluorescent Protein (RFP) in the first intron of Pax8 gene led to the expression of RFP in multiple tissues, including otic vesicles, midbrain-hindbrain boundary, spinal cord and kidney. The insertion also caused the inhibition of Pax8 expression. In Ikenaga et al. published in the Journal of Comparative Neurology, the role of Pax8 and its gene family member Pax2 was examined in the differentiation of spinal interneurons. In Nakayama et al. published in Development Growth and Differentiation, we combined the Pax8-RFP fish with another transgenic line, and examined the axon development of Pax8 expressing interneurons across the midline. In Diep et al. published in Nature, the expression of RFP in kidney was used to analyze the development of renal progenitor cells. In two papers, Won et al. published in the Journal of Neurophysiology and Won et al. published in PLoS ONE, we analyzed sodium and calcium channels expressed in primary sensory neurons, and studied their modifications by G-protein coupled receptors. We presented posters in several meetings including the Society for Neuroscience Meeting (Washington DC) and the International Conference on Zebrafish Development and Genetics (Madison, WI). Fumi Ono gave several invited talks at Universities (University of Massachusetts, Pennsylvania State University) and at international meetings (Neurotalk2012, Beijing, China;Ion Channel retreat, Vancouver, Canada;the International Conference on Zebrafish Development and Genetics, Madison, WI).

Project Start
Project End
Budget Start
Budget End
Support Year
Fiscal Year
Total Cost
Indirect Cost
National Institute on Alcohol Abuse and Alcoholism
Zip Code
Daikoku, Eriko; Saito, Masahisa; Ono, Fumihito (2015) Zebrafish mutants of the neuromuscular junction: swimming in the gene pool. J Physiol Sci 65:217-21
Puhl 3rd, Henry L; Lu, Van B; Won, Yu-Jin et al. (2014) Ancient origins of RGK protein function: modulation of voltage-gated calcium channels preceded the protostome and deuterostome split. PLoS One 9:e100694
Fero, Kandice; Bergeron, Sadie A; Horstick, Eric J et al. (2014) Impaired embryonic motility in dusp27 mutants reveals a developmental defect in myofibril structure. Dis Model Mech 7:289-98
Park, Jee-Young; Mott, Meghan; Williams, Tory et al. (2014) A single mutation in the acetylcholine receptor ?-subunit causes distinct effects in two types of neuromuscular synapses. J Neurosci 34:10211-8
Ikeda, Hiromi; Delargy, Alison H; Yokogawa, Tohei et al. (2013) Intrinsic properties of larval zebrafish neurons in ethanol. PLoS One 8:e63318
Ono, Fumihito (2013) Big answers from a small fly with a cultured brain. J Physiol 591:3
Nakayama, Sohei; Ikenaga, Takanori; Kawakami, Koichi et al. (2012) Transgenic line with gal4 insertion useful to study morphogenesis of craniofacial perichondrium, vascular endothelium-associated cells, floor plate, and dorsal midline radial glia during zebrafish development. Dev Growth Differ 54:202-15
Papke, Roger L; Ono, Fumihito; Stokes, Clare et al. (2012) The nicotinic acetylcholine receptors of zebrafish and an evaluation of pharmacological tools used for their study. Biochem Pharmacol 84:352-65
Walogorsky, Michael; Mongeon, Rebecca; Wen, Hua et al. (2012) Zebrafish model for congenital myasthenic syndrome reveals mechanisms causal to developmental recovery. Proc Natl Acad Sci U S A 109:17711-6
Park, Jee-Young; Ikeda, Hiromi; Ikenaga, Takanori et al. (2012) Acetylcholine receptors enable the transport of rapsyn from the Golgi complex to the plasma membrane. J Neurosci 32:7356-63

Showing the most recent 10 out of 16 publications