{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,17]],"date-time":"2026-10-17T07:38:46Z","timestamp":1792222726618,"version":"4.4.0"},"reference-count":59,"publisher":"The Company of Biologists","license":[{"start":{"date-parts":[[2021,5,18]],"date-time":"2021-05-18T00:00:00Z","timestamp":1621296000000},"content-version":"am","delay-in-days":2329,"URL":"http:\/\/creativecommons.org\/licenses\/by\/3.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2015,1,1]]},"abstract":"<jats:p>Plastic changes in synaptic properties are considered as fundamental for adaptive behaviors. Extracellular-signal-regulated kinase (ERK)-mediated signaling has been implicated in regulation of synaptic plasticity. Ribosomal S6 kinase 2 (RSK2) acts as a regulator and downstream effector of ERK. In the brain, RSK2 is predominantly expressed in regions required for learning and memory. Loss-of-function mutations in human RSK2 cause Coffin-Lowry Syndrome, which is characterized by severe mental retardation and low IQ scores in male patients. Knockout of RSK2 in mice or the RSK ortholog in Drosophila result in a variety of learning and memory defects. However, overall brain structure in these animals is not affected, leaving open the question of the pathophysiological consequences. Using the fly neuromuscular system as a model for excitatory glutamatergic synapses, we show that removal of RSK function causes distinct defects in motoneurons and at the neuromuscular junction. Based on histochemical and electrophysiological analyses we conclude that RSK is required for normal synaptic morphology and function. Furthermore, loss of RSK function interferes with ERK signaling at different levels. Elevated ERK activity was evident in the somata of motoneurons, whereas decreased ERK activity was observed in axons and the presynapse. In addition, we uncovered a novel function of RSK in anterograde axonal transport. Our results emphasize the importance of fine tuning ERK activity in neuronal processes underlying higher brain functions. In this context, RSK acts as a modulator of ERK signaling.<\/jats:p>","DOI":"10.1242\/dmm.021246","type":"journal-article","created":{"date-parts":[[2015,9,4]],"date-time":"2015-09-04T00:44:07Z","timestamp":1441327447000},"source":"Crossref","is-referenced-by-count":14,"title":["Loss of the Coffin-Lowry syndrome associated gene<i>RSK2<\/i>alters ERK activity, synaptic function and axonal transport in<i>Drosophila<\/i>motoneurons"],"prefix":"10.1242","author":[{"given":"Katherina","family":"Beck","sequence":"first","affiliation":[{"name":"University of W\u00fcrzburg, Institute of Medical Radiation and Cell Research, Versbacherstr. 5, D-97078 W\u00fcrzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nadine","family":"Ehmann","sequence":"additional","affiliation":[{"name":"University of W\u00fcrzburg, Institute of Physiology, Department of Neurophysiology, R\u00f6ntgenring 9, D-97070 W\u00fcrzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Till F.M.","family":"Andlauer","sequence":"additional","affiliation":[{"name":"University of W\u00fcrzburg, Rudolf Virchow Center, DFG Research Center for Experimental Biomedicine, Josef-Schneider-Str. 2, D-97080 W\u00fcrzburg, Germany; Freie Universit\u00e4t Berlin, Institute of Biology, Takusstr. 6, D-14195, Berlin, Germany; Max Planck Institute of Colloidals and Interfaces, Am M\u00fchlenberg 1, D-14476 Potsdam, Germany"},{"name":"Present address: Max Planck Institute of Psychiatry, Kraepelinstr. 2-10, 80804 M\u00fcnchen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dmitrij","family":"Ljaschenko","sequence":"additional","affiliation":[{"name":"University of W\u00fcrzburg, Institute of Physiology, Department of Neurophysiology, R\u00f6ntgenring 9, D-97070 W\u00fcrzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katrin","family":"Strecker","sequence":"additional","affiliation":[{"name":"University of W\u00fcrzburg, Institute of Medical Radiation and Cell Research, Versbacherstr. 5, D-97078 W\u00fcrzburg, Germany"},{"name":"Present address: German Center for Neurodegenerative Diseases (DZNE), Feodor-Lynen Str. 17, 81377 Munich, Germany; Institute for Metabolic Biochemistry, Ludwig-Maximilians University, Feodor-Lynen Str. 17, 81377 Munich, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthias","family":"Fischer","sequence":"additional","affiliation":[{"name":"University Hospital W\u00fcrzburg, Department of Psychiatry, Psychosomatics and Psychotherapy, F\u00fcchsleinstr. 15, 97080 W\u00fcrzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert J.","family":"Kittel","sequence":"additional","affiliation":[{"name":"University of W\u00fcrzburg, Institute of Physiology, Department of Neurophysiology, R\u00f6ntgenring 9, D-97070 W\u00fcrzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Raabe","sequence":"additional","affiliation":[{"name":"University of W\u00fcrzburg, Institute of Medical Radiation and Cell Research, Versbacherstr. 5, D-97078 W\u00fcrzburg, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2021042513063405800_DMM021246C1","doi-asserted-by":"publisher","first-page":"545","DOI":"10.1016\/S0896-6273(02)00589-5","article-title":"Wishful thinking encodes a BMP type II receptor that regulates synaptic growth in Drosophila","volume":"33","author":"Aberle","year":"2002","journal-title":"Neuron"},{"key":"2021042513063405800_DMM021246C2","doi-asserted-by":"publisher","first-page":"466","DOI":"10.1523\/JNEUROSCI.4034-08.2009","article-title":"Ribosomal S6 kinase cooperates with Casein kinase 2 to modulate the Drosophila circadian molecular oscillator","volume":"29","author":"Akten","year":"2009","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C3","doi-asserted-by":"publisher","first-page":"481","DOI":"10.1101\/pdb.prot068593","article-title":"In vivo imaging of the Drosophila larval neuromuscular junction","volume":"2012","author":"Andlauer","year":"2012","journal-title":"Cold Spring Harb. Protoc."},{"key":"2021042513063405800_DMM021246C4","doi-asserted-by":"publisher","first-page":"476","DOI":"10.1101\/pdb.prot068585","article-title":"Building an imaging chamber for in vivo imaging of Drosophila larvae","volume":"2012","author":"Andlauer","year":"2012","journal-title":"Cold Spring Harb. Protoc."},{"key":"2021042513063405800_DMM021246C5","doi-asserted-by":"publisher","first-page":"490","DOI":"10.1101\/pdb.prot068601","article-title":"Quantitative analysis of Drosophila larval neuromuscular junction morphology","volume":"2012","author":"Andlauer","year":"2012","journal-title":"Cold Spring Harb. Protoc."},{"key":"2021042513063405800_DMM021246C6","doi-asserted-by":"crossref","first-page":"1628","DOI":"10.1002\/j.1460-2075.1994.tb06426.x","article-title":"The Drosophila rolled locus encodes a MAP kinase required in the sevenless signal transduction pathway","volume":"13","author":"Biggs","year":"1994","journal-title":"EMBO J."},{"key":"2021042513063405800_DMM021246C7","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1242\/dev.118.2.401","article-title":"Targeted gene expression as a means of altering cell fates and generating dominant phenotypes","volume":"118","author":"Brand","year":"1993","journal-title":"Development"},{"key":"2021042513063405800_DMM021246C8","doi-asserted-by":"publisher","first-page":"1107","DOI":"10.3791\/1107","article-title":"Drosophila larval NMJ dissection","volume":"24","author":"Brent","year":"2009","journal-title":"J. Vis. Exp."},{"key":"2021042513063405800_DMM021246C9","doi-asserted-by":"publisher","first-page":"875","DOI":"10.1016\/0092-8674(94)90362-X","article-title":"A gain-of-function mutation in Drosophila MAP kinase activates multiple receptor tyrosine kinase pathways","volume":"76","author":"Brunner","year":"1994","journal-title":"Cell"},{"issue":"Suppl. 2","key":"2021042513063405800_DMM021246C10","doi-asserted-by":"publisher","first-page":"61","DOI":"10.1111\/j.1601-183X.2006.00230.x","article-title":"Mitogen-activated protein kinase\/extracellular regulated kinase signalling and memory stabilization: a review","volume":"5","author":"Davis","year":"2006","journal-title":"Genes Brain Behav."},{"key":"2021042513063405800_DMM021246C11","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1146\/annurev.neuro.31.061307.090711","article-title":"Role of axonal transport in neurodegenerative diseases","volume":"31","author":"De Vos","year":"2008","journal-title":"Annu. Rev. Neurosci."},{"key":"2021042513063405800_DMM021246C12","doi-asserted-by":"publisher","first-page":"348","DOI":"10.1016\/j.ydbio.2010.08.035","article-title":"Coffin-Lowry syndrome: a role for RSK2 in mammalian neurogenesis","volume":"347","author":"Dugani","year":"2010","journal-title":"Dev. Biol."},{"key":"2021042513063405800_DMM021246C13","doi-asserted-by":"publisher","first-page":"134","DOI":"10.1016\/j.mcn.2009.06.006","article-title":"P90 Ribosomal s6 kinase 2 negatively regulates axon growth in motoneurons","volume":"42","author":"Fischer","year":"2009","journal-title":"Mol. Cell. Neurosci."},{"key":"2021042513063405800_DMM021246C14","doi-asserted-by":"publisher","first-page":"212","DOI":"10.1002\/dneu.20700","article-title":"Drosophila RSK negatively regulates bouton number at the neuromuscular junction","volume":"69","author":"Fischer","year":"2009","journal-title":"Dev. Neurobiol."},{"key":"2021042513063405800_DMM021246C15","doi-asserted-by":"publisher","first-page":"3356","DOI":"10.1091\/mbc.E02-12-0800","article-title":"The kinesin-associated protein UNC-76 is required for axonal transport in the Drosophila nervous system","volume":"14","author":"Gindhart","year":"2003","journal-title":"Mol. Biol. Cell"},{"key":"2021042513063405800_DMM021246C16","doi-asserted-by":"publisher","first-page":"495","DOI":"10.1016\/j.conb.2008.10.003","article-title":"Axonal transport and the delivery of pre-synaptic components","volume":"18","author":"Goldstein","year":"2008","journal-title":"Curr. Opin. Neurobiol."},{"key":"2021042513063405800_DMM021246C17","doi-asserted-by":"publisher","first-page":"72","DOI":"10.2976\/1.3338710","article-title":"Mechanisms of short-term plasticity at neuromuscular active zones of Drosophila","volume":"4","author":"Hallermann","year":"2010","journal-title":"HFSP J."},{"key":"2021042513063405800_DMM021246C18","doi-asserted-by":"publisher","first-page":"610","DOI":"10.1016\/j.neuron.2010.09.039","article-title":"Molecular motors in neurons: transport mechanisms and roles in brain function, development, and disease","volume":"68","author":"Hirokawa","year":"2010","journal-title":"Neuron"},{"key":"2021042513063405800_DMM021246C19","doi-asserted-by":"publisher","first-page":"e1000193","DOI":"10.1371\/journal.pbio.1000193","article-title":"Negative regulation of active zone assembly by a newly identified SR protein kinase","volume":"7","author":"Johnson","year":"2009","journal-title":"PLoS Biol."},{"key":"2021042513063405800_DMM021246C20","doi-asserted-by":"publisher","first-page":"3056","DOI":"10.1038\/sj.emboj.7601180","article-title":"Inhibition of ERK-MAP kinase signaling by RSK during Drosophila development","volume":"25","author":"Kim","year":"2006","journal-title":"EMBO J."},{"key":"2021042513063405800_DMM021246C21","doi-asserted-by":"crossref","first-page":"2496","DOI":"10.1523\/JNEUROSCI.22-07-02496.2002","article-title":"The Ras1-mitogen-activated protein kinase signal transduction pathway regulates synaptic plasticity through Fasciclin II-mediated cell adhesion","volume":"22","author":"Koh","year":"2002","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C22","doi-asserted-by":"crossref","first-page":"4065","DOI":"10.1242\/dev.126.18.4065","article-title":"Development of the Drosophila mushroom bodies: sequential generation of three distinct types of neurons from a neuroblast","volume":"126","author":"Lee","year":"1999","journal-title":"Development"},{"key":"2021042513063405800_DMM021246C23","doi-asserted-by":"publisher","first-page":"8474","DOI":"10.1523\/JNEUROSCI.0409-14.2014","article-title":"Drosophila Syd-1, Liprin-alpha, and Protein Phosphatase 2A B\u2032 subunit Wrd function in a linear pathway to prevent ectopic accumulation of synaptic materials in distal axons","volume":"34","author":"Li","year":"2014","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C24","doi-asserted-by":"publisher","first-page":"2052","DOI":"10.1523\/JNEUROSCI.3278-10.2011","article-title":"Drosophila Acyl-CoA synthetase long-chain family member 4 regulates axonal transport of synaptic vesicles and is required for synaptic development and transmission","volume":"31","author":"Liu","year":"2011","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C25","doi-asserted-by":"publisher","first-page":"1407","DOI":"10.1016\/j.celrep.2013.04.003","article-title":"Hebbian plasticity guides maturation of glutamate receptor fields in vivo","volume":"3","author":"Ljaschenko","year":"2013","journal-title":"Cell Rep."},{"key":"2021042513063405800_DMM021246C26","doi-asserted-by":"publisher","first-page":"292","DOI":"10.1016\/j.neuron.2014.10.019","article-title":"Axonal transport: cargo-specific mechanisms of motility and regulation","volume":"84","author":"Maday","year":"2014","journal-title":"Neuron"},{"key":"2021042513063405800_DMM021246C27","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1016\/j.conb.2014.03.015","article-title":"Axon and dendritic trafficking","volume":"27","author":"Maeder","year":"2014","journal-title":"Curr. Opin. Neurobiol."},{"key":"2021042513063405800_DMM021246C28","doi-asserted-by":"publisher","first-page":"255","DOI":"10.1007\/s00439-010-0918-0","article-title":"Transcriptome profile reveals AMPA receptor dysfunction in the hippocampus of the Rsk2-knockout mice, an animal model of Coffin-Lowry syndrome","volume":"129","author":"Mehmood","year":"2011","journal-title":"Hum. Genet."},{"key":"2021042513063405800_DMM021246C29","doi-asserted-by":"publisher","first-page":"3358","DOI":"10.3390\/ijms14023358","article-title":"Rsk2 knockdown in PC12 cells results in Sp1 dependent increased expression of the Gria2 gene, encoding the AMPA receptor subunit GluR2","volume":"14","author":"Mehmood","year":"2013","journal-title":"Int. J. Mol. Sci."},{"key":"2021042513063405800_DMM021246C30","doi-asserted-by":"publisher","first-page":"647","DOI":"10.1002\/wdev.108","article-title":"Development and plasticity of the Drosophila larval neuromuscular junction","volume":"2","author":"Menon","year":"2013","journal-title":"Wiley Interdiscip. Rev. Dev. Biol."},{"key":"2021042513063405800_DMM021246C31","doi-asserted-by":"publisher","first-page":"281","DOI":"10.1093\/emboj\/21.3.281","article-title":"Glycogen synthase kinase 3 phosphorylates kinesin light chains and negatively regulates kinesin-based motility","volume":"21","author":"Morfini","year":"2002","journal-title":"EMBO J."},{"key":"2021042513063405800_DMM021246C32","doi-asserted-by":"publisher","first-page":"156","DOI":"10.1016\/j.nbd.2013.05.016","article-title":"Defective synaptic transmission and structure in the dentate gyrus and selective fear memory impairment in the Rsk2 mutant mouse model of Coffin-Lowry syndrome","volume":"58","author":"Morice","year":"2013","journal-title":"Neurobiol. Dis."},{"key":"2021042513063405800_DMM021246C33","doi-asserted-by":"publisher","first-page":"1244","DOI":"10.1038\/nature07003","article-title":"Analysis of a spatial orientation memory in Drosophila","volume":"453","author":"Neuser","year":"2008","journal-title":"Nature"},{"key":"2021042513063405800_DMM021246C34","doi-asserted-by":"publisher","first-page":"e1000700","DOI":"10.1371\/journal.pgen.1000700","article-title":"Bruchpilot in ribbon-like axonal agglomerates, behavioral defects, and early death in SRPK79D kinase mutants of Drosophila","volume":"5","author":"Nieratschker","year":"2009","journal-title":"PLoS Genet."},{"key":"2021042513063405800_DMM021246C35","doi-asserted-by":"publisher","first-page":"24939","DOI":"10.1074\/jbc.271.40.24939","article-title":"Biochemical characterization of RolledSem, an activated form of Drosophila mitogen-activated protein kinase","volume":"271","author":"Oellers","year":"1996","journal-title":"J. Biol. Chem."},{"key":"2021042513063405800_DMM021246C36","doi-asserted-by":"publisher","first-page":"1325","DOI":"10.1111\/j.1471-4159.2008.05703.x","article-title":"Dopaminergic system dysregulation in the mrsk2 KO mouse, an animal model of the Coffin-Lowry syndrome","volume":"107","author":"Pereira","year":"2008","journal-title":"J. Neurochem."},{"key":"2021042513063405800_DMM021246C37","doi-asserted-by":"publisher","first-page":"627","DOI":"10.1038\/ejhg.2009.189","article-title":"Coffin-Lowry syndrome","volume":"18","author":"Pereira","year":"2010","journal-title":"Eur. J. Hum. Genet."},{"key":"2021042513063405800_DMM021246C38","doi-asserted-by":"publisher","first-page":"2057","DOI":"10.1091\/mbc.E05-06-0526","article-title":"Kinesin-1 and Dynein are the primary motors for fast transport of mitochondria in Drosophila motor axons","volume":"17","author":"Pilling","year":"2006","journal-title":"Mol. Biol. Cell"},{"key":"2021042513063405800_DMM021246C39","doi-asserted-by":"publisher","first-page":"31","DOI":"10.1007\/s10519-006-9116-1","article-title":"Deletion of the Coffin\u2013Lowry Syndrome gene Rsk2 in mice is associated with impaired spatial learning and reduced control of exploratory behavior","volume":"37","author":"Poirier","year":"2007","journal-title":"Behav. Genet."},{"key":"2021042513063405800_DMM021246C40","doi-asserted-by":"publisher","first-page":"9745","DOI":"10.1523\/JNEUROSCI.3211-04.2004","article-title":"The S6KII (rsk) gene of Drosophila melanogaster differentially affects an operant and a classical learning task","volume":"24","author":"Putz","year":"2004","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C41","doi-asserted-by":"publisher","first-page":"3209","DOI":"10.1523\/JNEUROSCI.4194-04.2005","article-title":"Four different subunits are essential for expressing the synaptic glutamate receptor at neuromuscular junctions of Drosophila","volume":"25","author":"Qin","year":"2005","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C42","doi-asserted-by":"crossref","first-page":"3037","DOI":"10.1242\/dev.125.16.3037","article-title":"Divergent roles for NK-2 class homeobox genes in cardiogenesis in flies and mice","volume":"125","author":"Ranganayakulu","year":"1998","journal-title":"Development"},{"key":"2021042513063405800_DMM021246C43","doi-asserted-by":"publisher","first-page":"553","DOI":"10.1042\/BJ20110289","article-title":"Regulation and function of the RSK family of protein kinases","volume":"441","author":"Romeo","year":"2012","journal-title":"Biochem. J."},{"key":"2021042513063405800_DMM021246C44","doi-asserted-by":"publisher","first-page":"447","DOI":"10.1111\/j.1471-4159.2011.07423.x","article-title":"Altered ERK\/MAPK signaling in the hippocampus of the mrsk2_KO mouse model of Coffin-Lowry syndrome","volume":"119","author":"Schneider","year":"2011","journal-title":"J. Neurochem."},{"key":"2021042513063405800_DMM021246C45","doi-asserted-by":"publisher","first-page":"77","DOI":"10.1038\/nrn3156","article-title":"Mitochondrial transport in neurons: impact on synaptic homeostasis and neurodegeneration","volume":"13","author":"Sheng","year":"2012","journal-title":"Nat. Rev. Neurosci."},{"key":"2021042513063405800_DMM021246C46","doi-asserted-by":"publisher","first-page":"179","DOI":"10.1007\/BF00215114","article-title":"Improved stability of Drosophila larval neuromuscular preparations in haemolymph-like physiological solutions","volume":"175","author":"Stewart","year":"1994","journal-title":"J. Comp. Physiol. A"},{"key":"2021042513063405800_DMM021246C47","doi-asserted-by":"publisher","first-page":"1063","DOI":"10.1016\/S0896-6273(02)01094-2","article-title":"Axonal transport of mitochondria to synapses depends on milton, a novel Drosophila protein","volume":"36","author":"Stowers","year":"2002","journal-title":"Neuron"},{"key":"2021042513063405800_DMM021246C48","doi-asserted-by":"publisher","first-page":"16748","DOI":"10.1074\/jbc.M111.315929","article-title":"The C-terminal kinase and ERK-binding domains of Drosophila S6KII (RSK) are required for phosphorylation of the protein and modulation of circadian behavior","volume":"287","author":"Tangredi","year":"2012","journal-title":"J. Biol. Chem."},{"key":"2021042513063405800_DMM021246C49","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1038\/nrn1346","article-title":"MAPK cascade signalling and synaptic plasticity","volume":"5","author":"Thomas","year":"2004","journal-title":"Nat. Rev. Neurosci."},{"key":"2021042513063405800_DMM021246C50","doi-asserted-by":"publisher","first-page":"15006","DOI":"10.1073\/pnas.0507476102","article-title":"Ribosomal S6 kinase 2 interacts with and phosphorylates PDZ domain-containing proteins and regulates AMPA receptor transmission","volume":"102","author":"Thomas","year":"2005","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2021042513063405800_DMM021246C51","doi-asserted-by":"publisher","first-page":"3292","DOI":"10.1101\/gad.1734608","article-title":"UNC-51\/ATG1 kinase regulates axonal transport by mediating motor-cargo assembly","volume":"22","author":"Toda","year":"2008","journal-title":"Genes Dev."},{"key":"2021042513063405800_DMM021246C52","doi-asserted-by":"publisher","first-page":"431","DOI":"10.1038\/nmeth.1331","article-title":"Versatile P[acman] BAC libraries for transgenesis studies in Drosophila melanogaster","volume":"6","author":"Venken","year":"2009","journal-title":"Nat. Methods"},{"key":"2021042513063405800_DMM021246C53","doi-asserted-by":"publisher","first-page":"9293","DOI":"10.1523\/JNEUROSCI.1740-06.2006","article-title":"The B\u2032 protein phosphatase 2A regulatory subunit well-rounded regulates synaptic growth and cytoskeletal stability at the Drosophila neuromuscular junction","volume":"26","author":"Viquez","year":"2006","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C54","doi-asserted-by":"publisher","first-page":"833","DOI":"10.1016\/j.neuron.2006.02.008","article-title":"Bruchpilot, a protein with homology to ELKS\/CAST, is required for structural integrity and function of synaptic active zones in Drosophila","volume":"49","author":"Wagh","year":"2006","journal-title":"Neuron"},{"key":"2021042513063405800_DMM021246C55","doi-asserted-by":"publisher","first-page":"517","DOI":"10.1523\/JNEUROSCI.3848-08.2009","article-title":"Unc-51 controls active zone density and protein composition by downregulating ERK signaling","volume":"29","author":"Wairkar","year":"2009","journal-title":"J. Neurosci."},{"key":"2021042513063405800_DMM021246C56","doi-asserted-by":"publisher","first-page":"295","DOI":"10.1111\/tra.12037","article-title":"Endogenous GSK-3\/shaggy regulates bidirectional axonal transport of the amyloid precursor protein","volume":"14","author":"Weaver","year":"2013","journal-title":"Traffic"},{"key":"2021042513063405800_DMM021246C57","doi-asserted-by":"publisher","first-page":"7036","DOI":"10.1073\/pnas.92.15.7036","article-title":"Green fluorescent protein as a vital marker and reporter of gene expression in Drosophila","volume":"92","author":"Yeh","year":"1995","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2021042513063405800_DMM021246C58","doi-asserted-by":"publisher","first-page":"977","DOI":"10.1126\/science.8310297","article-title":"Paralysis and early death in cysteine string protein mutants of Drosophila","volume":"263","author":"Zinsmaier","year":"1994","journal-title":"Science"},{"key":"2021042513063405800_DMM021246C59","doi-asserted-by":"publisher","first-page":"107","DOI":"10.1007\/400_2009_20","article-title":"Mitochondrial transport dynamics in axons and dendrites","volume":"48","author":"Zinsmaier","year":"2009","journal-title":"Results Probl. Cell. Differ."}],"container-title":["Disease Models &amp; Mechanisms"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/journals.biologists.com\/dmm\/article-pdf\/8\/11\/1389\/1867773\/dmm021246.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.021246\/2069172\/dmm_021246v1.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"http:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.021246\/2069172\/dmm_021246v1.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,5,21]],"date-time":"2022-05-21T11:01:50Z","timestamp":1653130910000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dmm\/article\/doi\/10.1242\/dmm.021246\/257144\/Loss-of-the-Coffin-Lowry-syndrome-associated-gene"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,1,1]]},"references-count":59,"URL":"https:\/\/doi.org\/10.1242\/dmm.021246","relation":{},"ISSN":["1754-8411","1754-8403"],"issn-type":[{"value":"1754-8411","type":"electronic"},{"value":"1754-8403","type":"print"}],"subject":[],"published":{"date-parts":[[2015,1,1]]},"article-number":"dmm.021246"}}