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The Journal of Neuroscience, August 1, 2002, 22(15):6491-6498

The Zebrafish Motility Mutant twitch once Reveals New Roles for Rapsyn in Synaptic Function

Fumihito Ono¹, Anatoly Shcherbatko¹, Shin-ichi Higashijima², Gail Mandel², and Paul Brehm¹

¹ Department of Neurobiology and Behavior and ² Howard Hughes Medical Institute, State University of New York at Stony Brook, Stony Brook, New York 11794

Upon touch, twitch once zebrafish respond with one or two swimming strokes instead of typical full-blown escapes. This use-dependent fatigue is shown to be a consequence of a mutation in the tetratricopeptide domain of muscle rapsyn, inhibiting formation of subsynaptic acetylcholine receptor clusters. Physiological analysis indicates that reduced synaptic strength, attributable to loss of receptors, is augmented by a potent postsynaptic depression not seen at normal neuromuscular junctions. The synergism between these two physiological processes is causal to the use-dependent muscle fatigue. These findings offer insights into the physiological basis of human myasthenic syndrome and reveal the first demonstration of a role for rapsyn in regulating synaptic function.

Key words: tetratricopeptide repeats; synaptic depression; myasthenia gravis; synapse development; muscle fatigue; rapsyn

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Copyright © 2002 Society for Neuroscience  0270-6474/02/22156491-08$05.00/0

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