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In humans, the HSP-related atlastin genes ATL1\u2013ATL3 catalyze homotypic membrane fusion of endoplasmic reticulum (ER) tubules. How defects in neuronal Atlastin contribute to axonal degeneration has not been explained satisfactorily. Using Drosophila, we demonstrate that downregulation or overexpression of Atlastin in motor neurons results in decreased crawling speed and contraction frequency in larvae, while adult flies show progressive decline in climbing ability. Broad expression in the nervous system is required to rescue the atlastin-null Drosophila mutant (atl2) phenotype. Importantly, both spontaneous release and the reserve pool of synaptic vesicles are affected. Additionally, axonal secretory organelles are abnormally distributed, whereas presynaptic proteins diminish at terminals and accumulate in distal axons, possibly in lysosomes. Our findings suggest that trafficking defects produced by Atlastin dysfunction in motor neurons result in redistribution of presynaptic components and aberrant mobilization of synaptic vesicles, stressing the importance of ER-shaping proteins and the susceptibility of motor neurons to their mutations or depletion.<\/jats:p>","DOI":"10.1242\/jcs.201657","type":"journal-article","created":{"date-parts":[[2017,9,1]],"date-time":"2017-09-01T00:40:31Z","timestamp":1504226431000},"page":"3507-3516","source":"Crossref","is-referenced-by-count":30,"title":["<i>Drosophila<\/i> Atlastin in motor neurons is required for locomotion and presynaptic function"],"prefix":"10.1242","volume":"130","author":[{"given":"Cristian","family":"De Gregorio","sequence":"first","affiliation":[{"name":"Universidad de Chile 1 Department of Neuroscience , , Santiago CP8380453 , Chile"},{"name":"Biomedical Neuroscience Institute 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"},{"name":"Universidad de Chile 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ricardo","family":"Delgado","sequence":"additional","affiliation":[{"name":"Universidad de Chile 3 Department of Biology, Faculty of Sciences , , Santiago CP7800003 , Chile"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andr\u00e9s","family":"Ibacache","sequence":"additional","affiliation":[{"name":"Universidad de Chile 1 Department of Neuroscience , , Santiago CP8380453 , Chile"},{"name":"Biomedical Neuroscience Institute 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"},{"name":"Universidad de Chile 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jimena","family":"Sierralta","sequence":"additional","affiliation":[{"name":"Universidad de Chile 1 Department of Neuroscience , , Santiago CP8380453 , Chile"},{"name":"Biomedical Neuroscience Institute 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"},{"name":"Universidad de Chile 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4216-8672","authenticated-orcid":false,"given":"Andr\u00e9s","family":"Couve","sequence":"additional","affiliation":[{"name":"Universidad de Chile 1 Department of Neuroscience , , Santiago CP8380453 , Chile"},{"name":"Biomedical Neuroscience Institute 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"},{"name":"Universidad de Chile 2 , Faculty of Medicine , , Santiago CP8380453 , Chile"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2024061317120793100_JCS201657C1","doi-asserted-by":"publisher","first-page":"5091","DOI":"10.1073\/pnas.1217197110","article-title":"Inhibition of TFG function causes hereditary axon degeneration by impairing endoplasmic reticulum structure","volume":"110","author":"Beetz","year":"2013","journal-title":"Proc. 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