Genomes and Genes
Gene Symbol: Csp
Description: Cysteine string protein
Alias: CG6395, CSP, CSP32, Cys, D-CSP, D.M.CSP, DCSP-1, DNAJC5, DSP/DNAJC5, Dcsp, Dmel\CG6395, MAB ab49, ab49, anon-WO02059370.50, csp, csp29, csp32, l(3)03988, cysteine string protein, CG6395-PA, CG6395-PB, CG6395-PC, CG6395-PD, CG6395-PE, CG6395-PF, Csp-PA, Csp-PB, Csp-PC, Csp-PD, Csp-PE, Csp-PF, cystein string protein, cysteine string protein 3, cysteine-string protein
Species: fruit fly
- Fischer M, Raabe T, Heisenberg M, Sendtner M. Drosophila RSK negatively regulates bouton number at the neuromuscular junction. Dev Neurobiol. 2009;69:212-20 pubmed publisher..These experiments provided evidence that RSK mediates its negative effect on bouton formation at the Drosophila NMJ by inhibition of ERK signaling. ..
- Bate M, Broadie K. Wiring by fly: the neuromuscular system of the Drosophila embryo. Neuron. 1995;15:513-25 pubmed
- Razzaq A, Robinson I, McMahon H, Skepper J, Su Y, Zelhof A, et al. Amphiphysin is necessary for organization of the excitation-contraction coupling machinery of muscles, but not for synaptic vesicle endocytosis in Drosophila. Genes Dev. 2001;15:2967-79 pubmed..We therefore propose that muscle amphiphysin is not involved in clathrin-mediated endocytosis, but in the structural organization of the membrane-bound compartments of the excitation-contraction coupling machinery of muscles. ..
- Eberle K, Zinsmaier K, Buchner S, Gruhn M, Jenni M, Arnold C, et al. Wide distribution of the cysteine string proteins in Drosophila tissues revealed by targeted mutagenesis. Cell Tissue Res. 1998;294:203-17 pubmedThe "cysteine string protein" (CSP) genes of higher eukaryotes code for a novel family of proteins characterized by a "J" domain and an unusual cysteine-rich region...
- Bowman A, Kamal A, Ritchings B, Philp A, McGrail M, Gindhart J, et al. Kinesin-dependent axonal transport is mediated by the sunday driver (SYD) protein. Cell. 2000;103:583-94 pubmed..We propose that SYD mediates the axonal transport of at least one class of vesicles by interacting directly with KLC. ..
- Johnson E, Fetter R, Davis G. Negative regulation of active zone assembly by a newly identified SR protein kinase. PLoS Biol. 2009;7:e1000193 pubmed publisher..This mechanism could contribute to the speed and specificity with which AZs are assembled throughout the nervous system. ..
- Liebl F, Chen K, Karr J, Sheng Q, Featherstone D. Increased synaptic microtubules and altered synapse development in Drosophila sec8 mutants. BMC Biol. 2005;3:27 pubmed..We did not find any evidence that Sec8 is required for basal neurotransmission. ..
- Broadie K, Prokop A, Bellen H, O Kane C, Schulze K, Sweeney S. Syntaxin and synaptobrevin function downstream of vesicle docking in Drosophila. Neuron. 1995;15:663-73 pubmed..We conclude that the SNARE hypothesis cannot fully explain the role of these proteins in synaptic transmission. Instead, both proteins play distinct roles downstream of docking...
- Dawson Scully K, Lin Y, Imad M, Zhang J, Marin L, Horne J, et al. Morphological and functional effects of altered cysteine string protein at the Drosophila larval neuromuscular junction. Synapse. 2007;61:1-16 pubmedThe synaptic vesicle-associated cysteine string protein (CSP) is critical for neurotransmitter release at the neuromuscular junction (NMJ) of Drosophila, where the approximately 4% of mutant flies lacking CSP that survive to adulthood ..
- Gunawardena S, Goldstein L. Disruption of axonal transport and neuronal viability by amyloid precursor protein mutations in Drosophila. Neuron. 2001;32:389-401 pubmed..We discuss the possibility that axonal transport disruption may play a role in the neurodegenerative pathology of Alzheimer's disease. ..
- Arnold C, Reisch N, Leibold C, Becker S, Prüfert K, Sautter K, et al. Structure-function analysis of the cysteine string protein in Drosophila: cysteine string, linker and C terminus. J Exp Biol. 2004;207:1323-34 pubmed..genes coding for proteins with modified cysteine string, linker region or C terminus and transformed them into Csp null-mutant Drosophila: In the living animal, mutated CSP lacking all cysteines fails to associate with membranes, ..
- Mathew D, Gramates L, Packard M, Thomas U, Bilder D, Perrimon N, et al. Recruitment of scribble to the synaptic scaffolding complex requires GUK-holder, a novel DLG binding protein. Curr Biol. 2002;12:531-9 pubmed..Our results provide a mechanism by which developmentally important PDZ-mediated complexes are associated at the synapse. ..
- Fergestad T, Davis W, Broadie K. The stoned proteins regulate synaptic vesicle recycling in the presynaptic terminal. J Neurosci. 1999;19:5847-60 pubmed..These results suggest that the stoned proteins are essential for the recycling of synaptic vesicle membrane and are required for the proper sorting of synaptotagmin during endocytosis. ..
- McCabe B, Marques G, Haghighi A, Fetter R, Crotty M, Haerry T, et al. The BMP homolog Gbb provides a retrograde signal that regulates synaptic growth at the Drosophila neuromuscular junction. Neuron. 2003;39:241-54 pubmed..These experiments reveal that a muscle-derived BMP retrograde signal participates in coordinating neuromuscular synapse development and growth. ..
- Ranjan R, Bronk P, Zinsmaier K. Cysteine string protein is required for calcium secretion coupling of evoked neurotransmission in drosophila but not for vesicle recycling. J Neurosci. 1998;18:956-64 pubmedThe entire deletion of the cysteine string protein (CSP) gene causes a temperature-sensitive (ts) block of evoked neurotransmission in Drosophila...
- Gindhart J, Desai C, Beushausen S, Zinn K, Goldstein L. Kinesin light chains are essential for axonal transport in Drosophila. J Cell Biol. 1998;141:443-54 pubmed..Cell 64:1093-1102; Hurd, D.D., and W.M. Saxton. 1996. Genetics 144: 1075-1085) mutant phenotypes indicates that KLC is essential for kinesin function, perhaps by tethering KHC to intracellular cargos or by activating the kinesin motor. ..
- Bronk P, Wenniger J, Dawson Scully K, Guo X, Hong S, Atwood H, et al. Drosophila Hsc70-4 is critical for neurotransmitter exocytosis in vivo. Neuron. 2001;30:475-88 pubmedPrevious in vitro studies of cysteine-string protein (CSP) imply a potential role for the clathrin-uncoating ATPase Hsc70 in exocytosis...
- Zhang Y, Bailey A, Matthies H, Renden R, Smith M, Speese S, et al. Drosophila fragile X-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function. Cell. 2001;107:591-603 pubmed..dfxr futsch double mutants restore normal synaptic structure and function. We propose that dFXR acts as a translational repressor of Futsch to regulate microtubule-dependent synaptic growth and function. ..
- Koh T, Verstreken P, Bellen H. Dap160/intersectin acts as a stabilizing scaffold required for synaptic development and vesicle endocytosis. Neuron. 2004;43:193-205 pubmed..Our data suggest that Dap160, like dynamin, is involved in synaptic vesicle retrieval at active and periactive zones. ..
- Featherstone D, Davis W, Dubreuil R, Broadie K. Drosophila alpha- and beta-spectrin mutations disrupt presynaptic neurotransmitter release. J Neurosci. 2001;21:4215-24 pubmed
- McCabe B, Hom S, Aberle H, Fetter R, Marques G, Haerry T, et al. Highwire regulates presynaptic BMP signaling essential for synaptic growth. Neuron. 2004;41:891-905 pubmed..These results reveal a balance between positive BMP signaling and negative regulation by Highwire, governing the growth of neuromuscular synapses. ..
- Parfitt K, Reist N, Li J, Burgess R, Deitcher D, DiAntonio A, et al. Drosophila genetics and the functions of synaptic proteins. Cold Spring Harb Symp Quant Biol. 1995;60:371-7 pubmed
- Zelhof A, Bao H, Hardy R, Razzaq A, Zhang B, Doe C. Drosophila Amphiphysin is implicated in protein localization and membrane morphogenesis but not in synaptic vesicle endocytosis. Development. 2001;128:5005-15 pubmed..The strong misexpression phenotype coupled with the mild mutant and lack of phenotypes suggests that Amphiphysin acts redundantly with other proteins to organize specialized membrane domains within a diverse array of cell types. ..
- Dudu V, Bittig T, Entchev E, Kicheva A, Jülicher F, Gonzalez Gaitan M. Postsynaptic mad signaling at the Drosophila neuromuscular junction. Curr Biol. 2006;16:625-35 pubmed..We show that Mad nuclear trafficking depends on stimulation of the muscle. Our data suggest a mechanism linking synaptic transmission and postsynaptic TGF-beta signaling that may coordinate nerve-muscle development and function. ..
- Torroja L, Packard M, Gorczyca M, White K, Budnik V. The Drosophila beta-amyloid precursor protein homolog promotes synapse differentiation at the neuromuscular junction. J Neurosci. 1999;19:7793-803 pubmed..We propose a model by which APPL, in conjunction with activity-dependent mechanisms, regulates synaptic structure and number. ..
- Martin M, Iyadurai S, Gassman A, Gindhart J, Hays T, Saxton W. Cytoplasmic dynein, the dynactin complex, and kinesin are interdependent and essential for fast axonal transport. Mol Biol Cell. 1999;10:3717-28 pubmed..The shared bidirectional disruption phenotypes and the dominant genetic interactions demonstrate that cytoplasmic dynein, the dynactin complex, and conventional kinesin are interdependent in fast axonal transport. ..
- Zinsmaier K, Eberle K, Buchner E, Walter N, Benzer S. Paralysis and early death in cysteine string protein mutants of Drosophila. Science. 1994;263:977-80 pubmed..The csp gene of Drosophila encodes proteins homologous to synaptic vesicle proteins in Torpedo...
- Gonzalez Gaitan M, Jackle H. Role of Drosophila alpha-adaptin in presynaptic vesicle recycling. Cell. 1997;88:767-76 pubmed..Our results suggest an alpha-adaptin-dependent control of the vesicle cycle that maintains the balance between the amount of vesicle- and surface-associated membranes. ..
- Andrews H, Zhang Y, Trotta N, Broadie K. Drosophila sec10 is required for hormone secretion but not general exocytosis or neurotransmission. Traffic. 2002;3:906-21 pubmed..We conclude that dSec10 has no detectable role in most forms of polarized trafficking/exocytosis, including neurotransmission, but rather is essential for endocrine secretion. ..
- Verstreken P, Koh T, Schulze K, Zhai R, Hiesinger P, Zhou Y, et al. Synaptojanin is recruited by endophilin to promote synaptic vesicle uncoating. Neuron. 2003;40:733-48 pubmed..Our data also provide further evidence that kiss-and-run is able to maintain neurotransmitter release when synapses are not extensively challenged. ..
- Estes P, Roos J, van der Bliek A, Kelly R, Krishnan K, Ramaswami M. Traffic of dynamin within individual Drosophila synaptic boutons relative to compartment-specific markers. J Neurosci. 1996;16:5443-56 pubmed..We suggest that these dynamin-rich domains may mark the active zones for synaptic vesicle endocytosis first described at the frog neuromuscular junction. ..
- Beumer K, Rohrbough J, Prokop A, Broadie K. A role for PS integrins in morphological growth and synaptic function at the postembryonic neuromuscular junction of Drosophila. Development. 1999;126:5833-46 pubmed..These data provide the first genetic evidence for a functional role of integrins at the postembryonic synapse. ..
- Stimson D, Estes P, Rao S, Krishnan K, Kelly L, Ramaswami M. Drosophila stoned proteins regulate the rate and fidelity of synaptic vesicle internalization. J Neurosci. 2001;21:3034-44 pubmed..These observations are consistent with a model in which stonedA and stonedB act to facilitate the internalization of synaptic vesicle components from the plasma membrane. ..
- Zinsmaier K, Hofbauer A, Heimbeck G, Pflugfelder G, Buchner S, Buchner E. A cysteine-string protein is expressed in retina and brain of Drosophila. J Neurogenet. 1990;7:15-29 pubmedAntibodies can be used to identify tissue- and stage-specifically expressed genes. A monoclonal antibody MAB ab49 from a hybridoma library screened for immunohistochemical staining in the adult nervous system of Drosophila melanogaster ..
- Nie Z, Ranjan R, Wenniger J, Hong S, Bronk P, Zinsmaier K. Overexpression of cysteine-string proteins in Drosophila reveals interactions with syntaxin. J Neurosci. 1999;19:10270-9 pubmed..Here we show that the individual expression of various CSP isoforms in Drosophila similarly rescues the loss of evoked neurotransmitter release at csp null mutant motor nerve ..
- Guichet A, Wucherpfennig T, Dudu V, Etter S, Wilsch Bräuniger M, Hellwig A, et al. Essential role of endophilin A in synaptic vesicle budding at the Drosophila neuromuscular junction. EMBO J. 2002;21:1661-72 pubmed..Together with results of previous studies, these observations indicate that endophilin A is essential for synaptic vesicle endocytosis, being required from the onset of budding until fission. ..
- Fergestad T, Broadie K. Interaction of stoned and synaptotagmin in synaptic vesicle endocytosis. J Neurosci. 2001;21:1218-27 pubmed..We conclude that Stoned proteins control synaptic transmission strength by mediating the retrieval of Synaptotagmin I from the plasma membrane. ..
- Hiesinger P, Scholz M, Meinertzhagen I, Fischbach K, Obermayer K. Visualization of synaptic markers in the optic neuropils of Drosophila using a new constrained deconvolution method. J Comp Neurol. 2001;429:277-88 pubmed..We report differential localization of these near the resolution limit of light, which could not be distinguished without deconvolution. ..
- van de Goor J, Kelly R. Association of Drosophila cysteine string proteins with membranes. FEBS Lett. 1996;380:251-6 pubmed..This modification could be responsible for association of csp's with membranes. Extensive deacylation of Dcsp's by a 20 h incubation in 1 M hydroxylamine, pH 7...
- Song W, Ranjan R, Dawson Scully K, Bronk P, Marin L, Seroude L, et al. Presynaptic regulation of neurotransmission in Drosophila by the g protein-coupled receptor methuselah. Neuron. 2002;36:105-19 pubmed..Conditional expression of Mth restored normal release and normal vesicle docking and clustering but not the reduced size of synaptic sites, suggesting that Mth acutely adjusts vesicle trafficking to synaptic sites. ..
- Wagh D, Rasse T, Asan E, Hofbauer A, Schwenkert I, Dürrbeck H, et al. Bruchpilot, a protein with homology to ELKS/CAST, is required for structural integrity and function of synaptic active zones in Drosophila. Neuron. 2006;49:833-44 pubmed..We propose that BRP is critical for intact AZ structure and normal-evoked neurotransmitter release at chemical synapses of Drosophila. ..
- Murthy M, Garza D, Scheller R, Schwarz T. Mutations in the exocyst component Sec5 disrupt neuronal membrane traffic, but neurotransmitter release persists. Neuron. 2003;37:433-47 pubmed..In this regard, sec5 differs from the homologs of other yeast exocytosis genes that are required for both neuronal trafficking pathways. ..
- Verstreken P, Kjaerulff O, Lloyd T, Atkinson R, Zhou Y, Meinertzhagen I, et al. Endophilin mutations block clathrin-mediated endocytosis but not neurotransmitter release. Cell. 2002;109:101-12 pubmed..We propose that kiss-and-run maintains neurotransmission at active zones of the larval NMJ in endophilin animals. ..
- Reist N, Buchanan J, Li J, DiAntonio A, Buxton E, Schwarz T. Morphologically docked synaptic vesicles are reduced in synaptotagmin mutants of Drosophila. J Neurosci. 1998;18:7662-73 pubmed..The decreases in docking and evoked release are independent of the increase in spontaneous release. These results support the hypothesis that synaptotagmin stabilizes the docked state. ..
- Stimson D, Estes P, Smith M, Kelly L, Ramaswami M. A product of the Drosophila stoned locus regulates neurotransmitter release. J Neurosci. 1998;18:9638-49 pubmed..We propose a model in which stonedA functions together with synaptotagmin to regulate synaptic vesicle cycling. ..
- Rohrbough J, Pinto S, Mihalek R, Tully T, Broadie K. latheo, a Drosophila gene involved in learning, regulates functional synaptic plasticity. Neuron. 1999;23:55-70 pubmed..Our results suggest that LAT is a novel presynaptic protein with a role in the Ca2(+)-dependent synaptic modulation mechanisms necessary for behavioral plasticity. ..
- Haghnia M, Cavalli V, Shah S, Schimmelpfeng K, Brusch R, Yang G, et al. Dynactin is required for coordinated bidirectional motility, but not for dynein membrane attachment. Mol Biol Cell. 2007;18:2081-9 pubmed
- Daniels R, Collins C, Gelfand M, Dant J, Brooks E, Krantz D, et al. Increased expression of the Drosophila vesicular glutamate transporter leads to excess glutamate release and a compensatory decrease in quantal content. J Neurosci. 2004;24:10466-74 pubmed..These results demonstrate that (1) expression of DVGLUT determines the size and glutamate content of synaptic vesicles and (2) homeostatic mechanisms exist to attenuate the excitatory effects of excess glutamate release. ..
- Gunawardena S, Her L, Brusch R, Laymon R, Niesman I, Gordesky Gold B, et al. Disruption of axonal transport by loss of huntingtin or expression of pathogenic polyQ proteins in Drosophila. Neuron. 2003;40:25-40 pubmed..Thus, disruption of axonal transport by pathogenic polyQ proteins could contribute to early neuropathology in Huntington's and other polyQ expansion diseases. ..
- Bowman A, Patel King R, Benashski S, McCaffery J, Goldstein L, King S. Drosophila roadblock and Chlamydomonas LC7: a conserved family of dynein-associated proteins involved in axonal transport, flagellar motility, and mitosis. J Cell Biol. 1999;146:165-80 pubmed..We propose that roadblock/LC7 family members may modulate specific dynein functions. ..
- Pennetta G, Hiesinger P, Fabian Fine R, Meinertzhagen I, Bellen H. Drosophila VAP-33A directs bouton formation at neuromuscular junctions in a dosage-dependent manner. Neuron. 2002;35:291-306 pubmed..We propose that a DVAP-33A-mediated interaction between microtubules and presynaptic membrane plays a pivotal role during bouton budding. ..
- Ratnaparkhi A, Lawless G, Schweizer F, GOLSHANI P, Jackson G. A Drosophila model of ALS: human ALS-associated mutation in VAP33A suggests a dominant negative mechanism. PLoS ONE. 2008;3:e2334 pubmed publisher..This new fly model of ALS, with its robust pathological phenotypes, should for the first time allow the power of unbiased screens in Drosophila to be applied to study of motor neuron diseases. ..
- Bao H, Daniels R, MacLeod G, Charlton M, Atwood H, Zhang B. AP180 maintains the distribution of synaptic and vesicle proteins in the nerve terminal and indirectly regulates the efficacy of Ca2+-triggered exocytosis. J Neurophysiol. 2005;94:1888-903 pubmed..These results suggest that AP180 is required for either recycling vesicle proteins and/or maintaining the distribution of both vesicle and synaptic proteins in the nerve terminal. ..
- Featherstone D, Rushton E, Rohrbough J, Liebl F, Karr J, Sheng Q, et al. An essential Drosophila glutamate receptor subunit that functions in both central neuropil and neuromuscular junction. J Neurosci. 2005;25:3199-208 pubmed..These studies reveal GluRIID as a newly identified glutamate receptor subunit that is essential for glutamate receptor assembly/stabilization in the peripheral NMJ and required for properly patterned motor output in the CNS. ..
- Wu C, Wairkar Y, Collins C, DiAntonio A. Highwire function at the Drosophila neuromuscular junction: spatial, structural, and temporal requirements. J Neurosci. 2005;25:9557-66 pubmed
- Martin M, Ahern Djamali S, Hoffmann F, Saxton W. Abl tyrosine kinase and its substrate Ena/VASP have functional interactions with kinesin-1. Mol Biol Cell. 2005;16:4225-30 pubmed..A concomitant reduction in ena dosage rescued those defects. These results suggest that Ena/VASP, when not inhibited by the Abl pathway, can bind Khc and reduce its transport activity in axons. ..
- Prokop A, Landgraf M, Rushton E, Broadie K, Bate M. Presynaptic development at the Drosophila neuromuscular junction: assembly and localization of presynaptic active zones. Neuron. 1996;17:617-26 pubmed..Thus, the process of synapse formation can be genetically separated from the process of target recognition, revealing that localization of presynaptic active zones requires mef2-dependent muscle differentiation. ..
- Zhang Y, Rodesch C, Broadie K. Living synaptic vesicle marker: synaptotagmin-GFP. Genesis. 2002;34:142-5 pubmed