ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2012
Acetylcholine receptor gating in a zebrafish model for slow-channel syndrome.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 17 citations in OpenAlex.
- Article
- A mechano- and heat-gated two-pore domain KProceedings of the National Academy of Sciences of the United States of America · 2023Article
- Superfast excitation-contraction coupling in adult zebrafish skeletal muscle fibers.The Journal of general physiology · 2022Article
- Zebrafish neuromuscular junction: The power of N.Neuroscience letters · 2019Review
- Fatigue in Rapsyn-Deficient Zebrafish Reflects Defective Transmitter Release.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2016Article
- Zebrafish mutants of the neuromuscular junction: swimming in the gene pool.The journal of physiological sciences : JPS · 2015Review
- A single mutation in the acetylcholine receptor δ-subunit causes distinct effects in two types of neuromuscular synapses.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2014Article
- Neuromuscular disorders in zebrafish: state of the art and future perspectives.Neuromolecular medicine · 2013Review
- Zebrafish model for congenital myasthenic syndrome reveals mechanisms causal to developmental recovery.Proceedings of the National Academy of Sciences of the United States of America · 2012Article
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Authors and funding
5 authors at 2 institutions in 1 country.
Funding
Abstract
Slow-channel syndrome (SCS) is an autosomal-dominant disease resulting from mutations in muscle acetylcholine (ACh) receptor subunits. The associated fatigue and muscle degeneration are proposed to result from prolonged synaptic responses that overload intracellular calcium. Single-channel studies on reconstituted receptors bearing human mutations indicate that the prolonged responses result from an increase in receptor open duration and, in some cases, increased sensitivity to ACh. We show that both of these aberrant receptor properties are recapitulated in heterozygotic zebrafish bearing an L258P mutation in the α subunit, thus affording the unique opportunity to compare the single-channel properties of mutant receptors to the synaptic currents in vivo. Whole-cell recordings revealed synaptic currents that decayed along a multiexponential time course, reflecting receptors containing mixtures of wild-type and mutant α subunits. Treatment with quinidine, an open-channel blocker used to treat the human disorder, restored fast synaptic current kinetics and the ability to swim. Quinidine block also revealed that mutant receptors generate a large steady-state current in the absence of ACh. The spontaneous openings reflected a destabilization of the closed state, leading to an apparent increase in the sensitivity of these receptors to ACh. The effective block by quinidine on synaptic currents as well as nonliganded openings points to dual sources for the calcium-dependent myopathy in certain forms of SCS.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.