PUBLICATION
The zebrafish motility mutant twitch once reveals new roles for rapsyn in synaptic function
- Authors
- Ono, F., Shcherbatko, A., Higashijima, S.-I., Mandel, G., and Brehm, P.
- ID
- ZDB-PUB-020807-13
- Date
- 2002
- Source
- The Journal of neuroscience : the official journal of the Society for Neuroscience 22(15): 6491-6498 (Journal)
- Registered Authors
- Higashijima, Shin-ichi
- Keywords
- tetratricopeptide repeats; synaptic depression; myasthenia gravis; synapse development; muscle fatigue; rapsyn
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Behavior, Animal/physiology
- Electric Stimulation
- Escape Reaction/physiology
- Green Fluorescent Proteins
- Homozygote
- In Vitro Techniques
- Luminescent Proteins/genetics
- Membrane Potentials/physiology
- Motor Endplate/physiology
- Muscle Fatigue/genetics
- Muscle Fatigue/physiology
- Muscle Proteins/genetics
- Muscle Proteins/physiology*
- Muscle, Skeletal/innervation
- Muscle, Skeletal/metabolism
- Patch-Clamp Techniques
- Receptor Aggregation/physiology
- Receptors, Cholinergic/metabolism
- Recombinant Fusion Proteins/genetics
- Recombinant Fusion Proteins/metabolism
- Repetitive Sequences, Amino Acid/genetics
- Synapses/physiology*
- Zebrafish
- PubMed
- 12151528 Full text @ J. Neurosci.
Citation
Ono, F., Shcherbatko, A., Higashijima, S.-I., Mandel, G., and Brehm, P. (2002) The zebrafish motility mutant twitch once reveals new roles for rapsyn in synaptic function. The Journal of neuroscience : the official journal of the Society for Neuroscience. 22(15):6491-6498.
Abstract
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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping