{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T16:13:14Z","timestamp":1790439194279,"version":"4.1.0"},"reference-count":60,"publisher":"The Company of Biologists","issue":"4","license":[{"start":{"date-parts":[[2021,4,26]],"date-time":"2021-04-26T00:00:00Z","timestamp":1619395200000},"content-version":"vor","delay-in-days":25,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100015398","name":"Spinal Muscular Atrophy UK","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100015398","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100000406","name":"Motor Neurone Disease Association","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100000406","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Medical Research Council"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,4,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Synapses are particularly vulnerable in many neurodegenerative diseases and often the first to degenerate, for example in the motor neuron disease spinal muscular atrophy (SMA). Compounds that can counteract synaptic destabilisation are rare. Here, we describe an automated screening paradigm in zebrafish for small-molecule compounds that stabilize the neuromuscular synapse in vivo. We make use of a mutant for the axonal C-type lectin chondrolectin (chodl), one of the main genes dysregulated in SMA. In chodl\u2212\/\u2212 mutants, neuromuscular synapses that are formed at the first synaptic site by growing axons are not fully mature, causing axons to stall, thereby impeding further axon growth beyond that synaptic site. This makes axon length a convenient read-out for synapse stability. We screened 982 small-molecule compounds in chodl chodl\u2212\/\u2212 mutants and found four that strongly rescued motor axon length. Aberrant presynaptic neuromuscular synapse morphology was also corrected. The most-effective compound, the adenosine uptake inhibitor drug dipyridamole, also rescued axon growth defects in the UBA1-dependent zebrafish model of SMA. Hence, we describe an automated screening pipeline that can detect compounds with relevance to SMA. This versatile platform can be used for drug and genetic screens, with wider relevance to synapse formation and stabilisation.<\/jats:p>","DOI":"10.1242\/dmm.047761","type":"journal-article","created":{"date-parts":[[2021,4,27]],"date-time":"2021-04-27T03:14:20Z","timestamp":1619493260000},"source":"Crossref","is-referenced-by-count":18,"title":["Automated <i>in vivo<\/i> drug screen in zebrafish identifies synapse-stabilising drugs with relevance to spinal muscular atrophy"],"prefix":"10.1242","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9409-0830","authenticated-orcid":false,"given":"Ana-Maria","family":"Opri\u015foreanu","sequence":"first","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3670-5881","authenticated-orcid":false,"given":"Hannah L.","family":"Smith","sequence":"additional","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4700-5808","authenticated-orcid":false,"given":"Sophia","family":"Krix","sequence":"additional","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helena","family":"Chaytow","sequence":"additional","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"},{"name":"Euan MacDonald Centre for Motor Neurone Disease Research, University of Edinburgh, EH16 4SB Edinburgh, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5541-9747","authenticated-orcid":false,"given":"Neil O.","family":"Carragher","sequence":"additional","affiliation":[{"name":"Cancer Research UK Edinburgh Centre, MRC Institute of Genetics and Molecular Medicine, University of Edinburgh, EH4 2XR Edinburgh, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0306-5577","authenticated-orcid":false,"given":"Thomas H.","family":"Gillingwater","sequence":"additional","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"},{"name":"Euan MacDonald Centre for Motor Neurone Disease Research, University of Edinburgh, EH16 4SB Edinburgh, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9501-6165","authenticated-orcid":false,"given":"Catherina G.","family":"Becker","sequence":"additional","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"},{"name":"Euan MacDonald Centre for Motor Neurone Disease Research, University of Edinburgh, EH16 4SB Edinburgh, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2578-0819","authenticated-orcid":false,"given":"Thomas","family":"Becker","sequence":"additional","affiliation":[{"name":"Centre for Discovery Brain Sciences, University of Edinburgh, The Chancellor's Building, 49 Little France Crescent, Edinburgh EH16 4SB"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2021,4,26]]},"reference":[{"key":"2021082314153089200_DMM047761C1","doi-asserted-by":"publisher","first-page":"1073","DOI":"10.1016\/j.ajhg.2019.04.002","article-title":"Bi-allelic variants in DYNC1I2 cause syndromic microcephaly with intellectual disability, cerebral malformations, and dysmorphic facial features","volume":"104","author":"Ansar","year":"2019","journal-title":"Am. 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