TY - JOUR
T1 - Paralytic zebrafish lacking acetylcholine receptors fail to localize rapsyn clusters to the synapse
AU - Ono, Fumihito
AU - Higashijima, Shin Ichi
AU - Shcherbatko, Anatoly
AU - Fetcho, Joseph R.
AU - Brehm, Paul
PY - 2001/8/1
Y1 - 2001/8/1
N2 - Physiological analysis of two lines of paralytic mutant zebrafish, relaxed and sofa potato, reveals defects in distinct types of receptors in skeletal muscle. In sofa potato the paralysis results from failed synaptic transmission because of the absence of acetylcholine receptors, whereas relaxed mutants lack dihydropyridine receptor-mediated release of internal calcium in response to the muscle action potential. Synaptic structure and function appear normal in relaxed, showing that muscle paralysis per se does not impede proper synapse development. However, sofa potato mutants show incomplete development of the postsynaptic complex. Specifically, in the absence of ACh receptors, clusters of the receptor-aggregating protein rapsyn form in the extrasynaptic membrane but generally fail to localize to the subsynaptic region. Our results indicate that, although rapsyn molecules are capable of self-aggregation, interaction with ACh receptors is required for proper subsynaptic localization.
AB - Physiological analysis of two lines of paralytic mutant zebrafish, relaxed and sofa potato, reveals defects in distinct types of receptors in skeletal muscle. In sofa potato the paralysis results from failed synaptic transmission because of the absence of acetylcholine receptors, whereas relaxed mutants lack dihydropyridine receptor-mediated release of internal calcium in response to the muscle action potential. Synaptic structure and function appear normal in relaxed, showing that muscle paralysis per se does not impede proper synapse development. However, sofa potato mutants show incomplete development of the postsynaptic complex. Specifically, in the absence of ACh receptors, clusters of the receptor-aggregating protein rapsyn form in the extrasynaptic membrane but generally fail to localize to the subsynaptic region. Our results indicate that, although rapsyn molecules are capable of self-aggregation, interaction with ACh receptors is required for proper subsynaptic localization.
KW - Acetylcholine receptor
KW - Clustering
KW - Dihydropyridine receptor
KW - Mutant
KW - Neuromuscular junction
KW - Rapsyn
KW - Zebrafish
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U2 - 10.1523/jneurosci.21-15-05439.2001
DO - 10.1523/jneurosci.21-15-05439.2001
M3 - Article
C2 - 11466415
AN - SCOPUS:0035426034
SN - 0270-6474
VL - 21
SP - 5439
EP - 5448
JO - Journal of Neuroscience
JF - Journal of Neuroscience
IS - 15
ER -