chrna1

Summary

Gene Symbol: chrna1
Description: cholinergic receptor, nicotinic, alpha 1 (muscle)
Alias: nic1, zgc:86593, acetylcholine receptor subunit alpha, AChR alpha 1, cholinergic receptor, nicotinic, alpha polypeptide 1, nic, nic-1, nicotinic acetylcholine receptor alpha-1, nicotinic receptor
Species: zebrafish
Products:     chrna1

Top Publications

  1. Etard C, Behra M, Ertzer R, Fischer N, Jesuthasan S, Blader P, et al. Mutation in the delta-subunit of the nAChR suppresses the muscle defects caused by lack of Dystrophin. Dev Dyn. 2005;234:1016-25 pubmed
    ..The myofiber detachment phenotype of Dystroglycan-deficient embryos was not suppressed, suggesting that Dystrophin and Dystroglycan play distinct roles in muscle formation and maintenance of muscle integrity. ..
  2. Park J, Mott M, Williams T, Ikeda H, Wen H, Linhoff M, et al. A single mutation in the acetylcholine receptor ?-subunit causes distinct effects in two types of neuromuscular synapses. J Neurosci. 2014;34:10211-8 pubmed publisher
    ..Both wild-type and mutant larvae lacked ?/? subunits in slow muscle synapses. These findings in zebrafish suggest that some mutations in human congenital myasthenic syndromes may affect slow and fast muscle fibers differently. ..
  3. Ibanez Tallon I, Wen H, Miwa J, Xing J, Tekinay A, Ono F, et al. Tethering naturally occurring peptide toxins for cell-autonomous modulation of ion channels and receptors in vivo. Neuron. 2004;43:305-11 pubmed
    ..When combined with specific methods of cell and temporal expression, the extension of this approach to hundreds of naturally occurring peptide toxins opens a new landscape for cell-autonomous regulation of cellular physiology in vivo. ..
  4. Wen H, Linhoff M, McGinley M, Li G, Corson G, Mandel G, et al. Distinct roles for two synaptotagmin isoforms in synchronous and asynchronous transmitter release at zebrafish neuromuscular junction. Proc Natl Acad Sci U S A. 2010;107:13906-11 pubmed publisher
    ..Through these features, we further determined that during the height of shared synchronous and asynchronous transmission these two modes compete for the same release sites. ..
  5. Song Y, Selak M, Watson C, Coutts C, Scherer P, Panzer J, et al. Mechanisms underlying metabolic and neural defects in zebrafish and human multiple acyl-CoA dehydrogenase deficiency (MADD). PLoS ONE. 2009;4:e8329 pubmed publisher
    ..These results show that mitochondrial dysfunction, leading to an increase in aerobic glycolysis, affects neurogenesis through the PPARG-ERK pathway, a potential target for therapeutic intervention. ..
  6. Benedetti L, Ghilardi A, Rottoli E, De Maglie M, Prosperi L, Perego C, et al. INaP selective inhibition reverts precocious inter- and motorneurons hyperexcitability in the Sod1-G93R zebrafish ALS model. Sci Rep. 2016;6:24515 pubmed publisher
    ..Our findings provide a valid phenotype-based tool for unbiased in vivo drug screening that can be used to develop new therapies. ..
  7. Walogorsky M, Mongeon R, Wen H, Nelson N, Urban J, Ono F, et al. Zebrafish model for congenital myasthenic syndrome reveals mechanisms causal to developmental recovery. Proc Natl Acad Sci U S A. 2012;109:17711-6 pubmed publisher
    ..Thus, the continued expression of ?(twi) into adulthood is tolerated because of the ? expression and associated recovery, raising the likelihood of unappreciated myasthenic cases that benefit from the ?-? switch. ..
  8. Sepich D, Ho R, Westerfield M. Autonomous expression of the nic1 acetylcholine receptor mutation in zebrafish muscle cells. Dev Biol. 1994;161:84-90 pubmed
    The nic1b107 (nic1) mutation blocks expression of both functional and clustered acetylcholine receptors (AChRs) in zebrafish muscle. Normally, signaling between motoneurons and muscles regulates AChR clustering...
  9. Gupta V, Kawahara G, Gundry S, Chen A, Lencer W, Zhou Y, et al. The zebrafish dag1 mutant: a novel genetic model for dystroglycanopathies. Hum Mol Genet. 2011;20:1712-25 pubmed publisher

More Information

Publications31

  1. Shwartz Y, Farkas Z, Stern T, Aszodi A, Zelzer E. Muscle contraction controls skeletal morphogenesis through regulation of chondrocyte convergent extension. Dev Biol. 2012;370:154-63 pubmed publisher
  2. Petzold A, Balciunas D, Sivasubbu S, Clark K, Bedell V, Westcot S, et al. Nicotine response genetics in the zebrafish. Proc Natl Acad Sci U S A. 2009;106:18662-7 pubmed publisher
  3. Jing L, Lefebvre J, Gordon L, Granato M. Wnt signals organize synaptic prepattern and axon guidance through the zebrafish unplugged/MuSK receptor. Neuron. 2009;61:721-33 pubmed publisher
    ..We propose that Wnt ligands activate unplugged/MuSK signaling in muscle fibers to restrict growth cone guidance and AChR prepatterns to the muscle center through a mechanism reminiscent of the planar cell polarity pathway. ..
  4. Lefebvre J, Ono F, Puglielli C, Seidner G, Franzini Armstrong C, Brehm P, et al. Increased neuromuscular activity causes axonal defects and muscular degeneration. Development. 2004;131:2605-18 pubmed
    ..a novel, dominant gain-of-function mutation in the muscle-specific nicotinic acetylcholine receptor alpha-subunit, CHRNA1. Moreover, electrophysiological analysis demonstrates that the mutant subunit increases synaptic decay times, ..
  5. Guillon E, Bretaud S, Ruggiero F. Slow Muscle Precursors Lay Down a Collagen XV Matrix Fingerprint to Guide Motor Axon Navigation. J Neurosci. 2016;36:2663-76 pubmed publisher
    ..Zebrafish and humans use common molecular cues and regulatory mechanisms for the neuromuscular system development. And as such, our study reveals COL15A1 as a candidate gene for orphan neuromuscular disorders. ..
  6. Papke R, Ono F, Stokes C, Urban J, Boyd R. The nicotinic acetylcholine receptors of zebrafish and an evaluation of pharmacological tools used for their study. Biochem Pharmacol. 2012;84:352-65 pubmed publisher
    ..Our data therefore indicate that while some pharmacological tools used in zebrafish may function as expected, others will not. ..
  7. Maves L, Waskiewicz A, Paul B, Cao Y, Tyler A, Moens C, et al. Pbx homeodomain proteins direct Myod activity to promote fast-muscle differentiation. Development. 2007;134:3371-82 pubmed
    ..Our results reveal that Pbx proteins modulate Myod activity to drive fast-muscle gene expression, thus showing that homeodomain proteins can direct bHLH proteins to establish a specific cell-type identity. ..
  8. Brennan C, Mangoli M, Dyer C, Ashworth R. Acetylcholine and calcium signalling regulates muscle fibre formation in the zebrafish embryo. J Cell Sci. 2005;118:5181-90 pubmed
  9. Ahmed K, Ali D. Nicotinic acetylcholine receptors (nAChRs) at zebrafish red and white muscle show different properties during development. Dev Neurobiol. 2016;76:916-36 pubmed publisher
    ..RT-qPCR of mRNA for nicotinic receptor subunits supports a switch from ? to ? subunits in white fibers but not in red...
  10. Sun J, Pan C, Chew T, Liang F, Burmeister M, Low B. BNIP-H Recruits the Cholinergic Machinery to Neurite Terminals to Promote Acetylcholine Signaling and Neuritogenesis. Dev Cell. 2015;34:555-68 pubmed publisher
    ..Therefore, precise targeting of the cholinergic machinery through BNIP-H is essential for the local production of ACh for morphogenesis and neurotransmission. ..
  11. Huang L, Zuo Z, Zhang Y, Wu M, Lin J, Wang C. Use of toxicogenomics to predict the potential toxic effect of Benzo(a)pyrene on zebrafish embryos: ocular developmental toxicity. Chemosphere. 2014;108:55-61 pubmed publisher
    ..Our results were helpful for an understanding of the toxicity of BaP. This study also indicated that microarray analysis was effective for predicting the potential toxicity of chemicals with high sensitivity and accuracy. ..
  12. Menelaou E, Udvadia A, Tanguay R, Svoboda K. Activation of ?2A-containing nicotinic acetylcholine receptors mediates nicotine-induced motor output in embryonic zebrafish. Eur J Neurosci. 2014;40:2225-40 pubmed publisher
    ..These findings also indicate that functional neuronal nAChRs are present within spinal cord at the time when locomotor output in zebrafish first begins to manifest itself. ..
  13. Brent L, Drapeau P. Targeted "knockdown" of channel expression in vivo with an antisense morpholino oligonucleotide. Neuroscience. 2002;114:275-8 pubmed
    ..Because of their stability and specificity, morpholinos should prove useful for targeted deletion of transmitter receptors and channels in developing zebrafish and possibly in other preparations. ..
  14. Fero K, Bergeron S, Horstick E, Codore H, Li G, Ono F, et al. Impaired embryonic motility in dusp27 mutants reveals a developmental defect in myofibril structure. Dis Model Mech. 2014;7:289-98 pubmed publisher
    ..These findings are the first to implicate a functional role of dusp27 as a gene required for myofiber maturation and provide an animal model for analyzing the mechanisms governing myofibril assembly. ..
  15. Wei C, Thatcher E, Olena A, Cha D, Perdigoto A, Marshall A, et al. miR-153 regulates SNAP-25, synaptic transmission, and neuronal development. PLoS ONE. 2013;8:e57080 pubmed publisher
    ..Together, our results indicate that precise control of SNAP-25 expression by miR-153 is critically important for proper neuronal patterning as well as neurotransmission. ..
  16. Walogorsky M, Mongeon R, Wen H, Mandel G, Brehm P. Acetylcholine receptor gating in a zebrafish model for slow-channel syndrome. J Neurosci. 2012;32:7941-8 pubmed publisher
    ..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. ..
  17. Mongeon R, Walogorsky M, Urban J, Mandel G, Ono F, Brehm P. An acetylcholine receptor lacking both ? and ? subunits mediates transmission in zebrafish slow muscle synapses. J Gen Physiol. 2011;138:353-66 pubmed publisher
    ..The unique functional features offered by the ?(2)??(2) receptor likely play a central role in mediating the persistent contractions characteristic to this muscle type. ..
  18. Mackay E, Apschner A, Schulte Merker S. Vitamin K reduces hypermineralisation in zebrafish models of PXE and GACI. Development. 2015;142:1095-101 pubmed publisher
    ..Additionally, we found that abcc6a is strongly expressed at the site of mineralisation rather than the liver, as it is in mammals, which has significant implications for our understanding of the function of ABCC6. ..
  19. EPLEY K, Urban J, Ikenaga T, Ono F. A modified acetylcholine receptor delta-subunit enables a null mutant to survive beyond sexual maturation. J Neurosci. 2008;28:13223-31 pubmed publisher
    ..In the rescued fish, a foreign promoter drove the transgene expression and the NMJ had altered synaptic strength. The survival of the transgenic animal delineates requirements for gene therapies of NMJ. ..
  20. Ramesh T, Lyon A, Pineda R, Wang C, Janssen P, Canan B, et al. A genetic model of amyotrophic lateral sclerosis in zebrafish displays phenotypic hallmarks of motoneuron disease. Dis Model Mech. 2010;3:652-62 pubmed publisher
    ..These phenotypes were more severe in lines expressing higher levels of mutant Sod1 and were absent in lines overexpressing wild-type Sod1. Thus, we have generated a vertebrate model of ALS to complement existing mammal models...
  21. Muller J, Jepson C, Laval S, Bushby K, Straub V, Lochmuller H. Dok-7 promotes slow muscle integrity as well as neuromuscular junction formation in a zebrafish model of congenital myasthenic syndromes. Hum Mol Genet. 2010;19:1726-40 pubmed publisher
    ..Our findings in the zebrafish model contribute to a better understanding of the signalling pathways at the NMJ and the pathomechanisms of DOK7 CMSs. ..
  22. Boon K, Xiao S, McWhorter M, Donn T, Wolf Saxon E, Bohnsack M, et al. Zebrafish survival motor neuron mutants exhibit presynaptic neuromuscular junction defects. Hum Mol Genet. 2009;18:3615-25 pubmed publisher
    ..Our analysis indicates the requirement for Smn in motoneurons to maintain SV2 in presynaptic terminals indicating that Smn, either directly or indirectly, plays a role in presynaptic integrity. ..