{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,12]],"date-time":"2026-03-12T09:39:50Z","timestamp":1773308390867,"version":"3.50.1"},"reference-count":48,"publisher":"SAGE Publications","issue":"1","license":[{"start":{"date-parts":[[2018,10,27]],"date-time":"2018-10-27T00:00:00Z","timestamp":1540598400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2018,10,27]],"date-time":"2018-10-27T00:00:00Z","timestamp":1540598400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Zebrafish"],"published-print":{"date-parts":[[2019,2,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>\n                    Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease characterized by progressive loss of motor neurons. ALS can be modeled in zebrafish (\n                    <jats:italic toggle=\"yes\">Danio rerio<\/jats:italic>\n                    ) through the expression of human ALS-causing genes, such as superoxide dismutase 1 (\n                    <jats:italic toggle=\"yes\">SOD1<\/jats:italic>\n                    ). Overexpression of mutated human SOD1 protein causes aberrant branching and shortening of spinal motor axons. Despite this, the functional relevance of this axon morphology remains elusive. Our aim was to determine whether this motor axonopathy is correlated with impaired movement in mutant (MT) SOD1-expressing zebrafish. Transgenic zebrafish embryos that express blue fluorescent protein (mTagBFP) in motor neurons were injected with either wild-type (WT) or MT (A4V) human SOD1 messenger ribonucleic acid (mRNA). At 48 hours post-fertilization, larvae movement (distance traveled during behavioral testing) was examined, followed by quantification of motor axon length. Larvae injected with MT SOD1 mRNA had significantly shorter and more aberrantly branched motor axons (\n                    <jats:italic toggle=\"yes\">p<\/jats:italic>\n                    \u2009&lt;\u20090.002) and traveled a significantly shorter distance during behavioral testing (\n                    <jats:italic toggle=\"yes\">p<\/jats:italic>\n                    \u2009&lt;\u20090.001) when compared with WT SOD1 and noninjected larvae. Furthermore, there was a positive correlation between distance traveled and motor axon length (\n                    <jats:italic toggle=\"yes\">R<\/jats:italic>\n                    <jats:sup>2<\/jats:sup>\n                    \u2009=\u20090.357,\n                    <jats:italic toggle=\"yes\">p<\/jats:italic>\n                    \u2009&lt;\u20090.001). These data represent the first correlative investigation of motor axonopathies and impaired movement in SOD1-expressing zebrafish, confirming functional relevance and validating movement as a disease phenotype for the testing of disease treatments for ALS.\n                  <\/jats:p>","DOI":"10.1089\/zeb.2018.1588","type":"journal-article","created":{"date-parts":[[2018,10,9]],"date-time":"2018-10-09T17:14:54Z","timestamp":1539105294000},"page":"8-14","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":25,"title":["Motor Neuron Abnormalities Correlate with Impaired Movement in Zebrafish that Express Mutant Superoxide Dismutase 1"],"prefix":"10.1177","volume":"16","author":[{"given":"Katherine J.","family":"Robinson","sequence":"first","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kristy C.","family":"Yuan","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Emily K.","family":"Don","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alison L.","family":"Hogan","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Claire G.","family":"Winnick","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Madelaine C.","family":"Tym","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Caitlin W.","family":"Lucas","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hamideh","family":"Shahheydari","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maxinne","family":"Watchon","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."},{"name":"Sydney Medical School, University of Sydney, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ian P.","family":"Blair","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Julie D.","family":"Atkin","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Garth A.","family":"Nicholson","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."},{"name":"Concord Clinical School and ANZAC Research Institute, Concord Repatriation Hospital, Concord, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicholas J.","family":"Cole","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, Faculty of Medicine and Health Sciences, Centre for Motor Neuron Disease Research, Macquarie University, Sydney, Australia."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Angela S.","family":"Laird","sequence":"additional","affiliation":[{"name":"Department of 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