{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T09:45:57Z","timestamp":1791279957833,"version":"4.1.0"},"reference-count":73,"publisher":"Rockefeller University Press","issue":"5","content-domain":{"domain":["rupress.org"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2003,9,1]]},"abstract":"<jats:p>Spinal muscular atrophy (SMA) is an autosomal recessive disorder characterized by a loss of \u03b1 motoneurons in the spinal cord. SMA is caused by low levels of the ubiquitously expressed survival motor neuron (Smn) protein. As it is unclear how low levels of Smn specifically affect motoneurons, we have modeled SMA in zebrafish, a vertebrate model organism with well-characterized motoneuron development. Using antisense morpholinos to reduce Smn levels throughout the entire embryo, we found motor axon\u2013specific pathfinding defects. Reduction of Smn in individual motoneurons revealed that smn is acting cell autonomously. These results show for the first time, in vivo, that Smn functions in motor axon development and suggest that these early developmental defects may lead to subsequent motoneuron loss.<\/jats:p>","DOI":"10.1083\/jcb.200303168","type":"journal-article","created":{"date-parts":[[2003,9,2]],"date-time":"2003-09-02T20:37:03Z","timestamp":1062535023000},"page":"919-932","update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":386,"title":["Knockdown of the survival motor neuron (Smn) protein in zebrafish causes defects in motor axon outgrowth and pathfinding"],"prefix":"10.1083","volume":"162","author":[{"given":"Michelle L.","family":"McWhorter","sequence":"first","affiliation":[{"name":"1Center for Molecular Neurobiology, Cellular, and Developmental Biology Program"},{"name":"2Molecular, Cellular, and Developmental Biology Program"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Umrao R.","family":"Monani","sequence":"additional","affiliation":[{"name":"3Department of Neurology, The Ohio State University, Columbus, OH 43210"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Arthur H.M.","family":"Burghes","sequence":"additional","affiliation":[{"name":"2Molecular, Cellular, and Developmental Biology Program"},{"name":"3Department of Neurology, The Ohio State University, Columbus, OH 43210"},{"name":"4Department of Molecular and Cellular Biochemistry, The Ohio State University, Columbus, OH 43210"},{"name":"5Department of Molecular Genetics, The Ohio State University, Columbus, OH 43210"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine E.","family":"Beattie","sequence":"additional","affiliation":[{"name":"1Center for Molecular Neurobiology, Cellular, and Developmental Biology Program"},{"name":"2Molecular, Cellular, and Developmental Biology Program"},{"name":"6Department of Neuroscience, The Ohio State University, Columbus, OH 43210"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2003,9,2]]},"reference":[{"key":"2023072214412877100_BIB1","doi-asserted-by":"crossref","first-page":"4117","DOI":"10.1242\/dev.121.12.4117","volume":"121","year":"1995","journal-title":"Development."},{"key":"2023072214412877100_BIB2","doi-asserted-by":"crossref","first-page":"489","DOI":"10.1016\/S0361-9230(00)00382-8","volume":"53","year":"2000","journal-title":"Brain Res. 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