{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T18:06:54Z","timestamp":1791396414642,"version":"4.3.3"},"reference-count":72,"publisher":"Society for Neuroscience","issue":"44","license":[{"start":{"date-parts":[[2015,4,29]],"date-time":"2015-04-29T00:00:00Z","timestamp":1430265600000},"content-version":"vor","delay-in-days":182,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-sa\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. Neurosci."],"published-print":{"date-parts":[[2014,10,29]]},"abstract":"<jats:p>Mutations in Kinesin proteins (Kifs) are linked to various neurological diseases, but the specific and redundant functions of the vertebrate Kifs are incompletely understood. For example, Kif5A, but not other Kinesin-1 heavy-chain family members, is implicated in Charcot-Marie-Tooth disease (CMT) and Hereditary Spastic Paraplegia (HSP), but the mechanism of its involvement in the progressive axonal degeneration characteristic of these diseases is not well understood. We report that zebrafish<jats:italic>kif5Aa<\/jats:italic>mutants exhibit hyperexcitability, peripheral polyneuropathy, and axonal degeneration reminiscent of CMT and HSP. Strikingly, although<jats:italic>kif5<\/jats:italic>genes are thought to act largely redundantly in other contexts, and zebrafish peripheral neurons express five<jats:italic>kif5<\/jats:italic>genes,<jats:italic>kif5Aa<\/jats:italic>mutant peripheral sensory axons lack mitochondria and degenerate. We show that this Kif5Aa-specific function is cell autonomous and is mediated by its C-terminal tail, as only Kif5Aa and chimeric motors containing the Kif5Aa C-tail can rescue deficits. Finally, concurrent loss of the<jats:italic>kinesin-3<\/jats:italic>,<jats:italic>kif1b<\/jats:italic>, or its adaptor<jats:italic>kbp<\/jats:italic>, exacerbates axonal degeneration via a nonmitochondrial cargo common to Kif5Aa. Our results shed light on Kinesin complexity and reveal determinants of specific Kif5A functions in mitochondrial transport, adaptor binding, and axonal maintenance.<\/jats:p>","DOI":"10.1523\/jneurosci.2770-14.2014","type":"journal-article","created":{"date-parts":[[2014,10,29]],"date-time":"2014-10-29T17:07:51Z","timestamp":1414602471000},"page":"14717-14732","source":"Crossref","is-referenced-by-count":120,"title":["Unique Function of Kinesin Kif5A in Localization of Mitochondria in Axons"],"prefix":"10.1523","volume":"34","author":[{"given":"Philip D.","family":"Campbell","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kimberle","family":"Shen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew R.","family":"Sapio","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas D.","family":"Glenn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William S.","family":"Talbot","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9362-0342","authenticated-orcid":false,"given":"Florence L.","family":"Marlow","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"393","published-online":{"date-parts":[[2014,10,29]]},"reference":[{"key":"2023041304134991000_34.44.14717.1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0054166"},{"key":"2023041304134991000_34.44.14717.2","doi-asserted-by":"publisher","DOI":"10.1038\/nmeth.2434"},{"key":"2023041304134991000_34.44.14717.3","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2601-12.2012"},{"key":"2023041304134991000_34.44.14717.4","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroscience.2004.11.031"},{"key":"2023041304134991000_34.44.14717.5","doi-asserted-by":"publisher","DOI":"10.1111\/j.1528-1167.2007.01075.x"},{"key":"2023041304134991000_34.44.14717.6","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.010090"},{"key":"2023041304134991000_34.44.14717.7","doi-asserted-by":"publisher","DOI":"10.1016\/0143-4160(88)90036-X"},{"key":"2023041304134991000_34.44.14717.8","doi-asserted-by":"publisher","DOI":"10.1007\/s10048-005-0027-8"},{"key":"2023041304134991000_34.44.14717.9","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M111.236018"},{"key":"2023041304134991000_34.44.14717.10","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M409095200"},{"key":"2023041304134991000_34.44.14717.11","doi-asserted-by":"crossref","first-page":"197","DOI":"10.1152\/jn.2001.86.1.197","article-title":"Synaptic drive to motoneurons during fictive swimming in the developing zebrafish","volume":"86","author":"Buss","year":"2001","journal-title":"J Neurophysiol"},{"key":"2023041304134991000_34.44.14717.12","doi-asserted-by":"publisher","DOI":"10.1016\/j.gep.2013.05.002"},{"key":"2023041304134991000_34.44.14717.13","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201302040"},{"key":"2023041304134991000_34.44.14717.14","doi-asserted-by":"publisher","DOI":"10.1038\/nrn1788"},{"key":"2023041304134991000_34.44.14717.15","doi-asserted-by":"publisher","DOI":"10.1038\/13001"},{"key":"2023041304134991000_34.44.14717.16","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2011.01717.x"},{"key":"2023041304134991000_34.44.14717.17","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2990.2004.00587.x"},{"key":"2023041304134991000_34.44.14717.18","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1003303"},{"key":"2023041304134991000_34.44.14717.19","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000138731.60693.D2"},{"key":"2023041304134991000_34.44.14717.20","doi-asserted-by":"publisher","DOI":"10.1016\/j.jns.2012.03.025"},{"key":"2023041304134991000_34.44.14717.21","doi-asserted-by":"publisher","DOI":"10.1038\/13008"},{"key":"2023041304134991000_34.44.14717.22","doi-asserted-by":"publisher","DOI":"10.1002\/humu.20920"},{"key":"2023041304134991000_34.44.14717.23","doi-asserted-by":"publisher","DOI":"10.1038\/35010525"},{"key":"2023041304134991000_34.44.14717.24","doi-asserted-by":"publisher","DOI":"10.1021\/bi8022575"},{"key":"2023041304134991000_34.44.14717.25","doi-asserted-by":"publisher","DOI":"10.1101\/gr.143000"},{"key":"2023041304134991000_34.44.14717.26","doi-asserted-by":"publisher","DOI":"10.1007\/s00234-005-1415-3"},{"key":"2023041304134991000_34.44.14717.27","doi-asserted-by":"publisher","DOI":"10.1016\/j.tcb.2013.08.007"},{"key":"2023041304134991000_34.44.14717.28","doi-asserted-by":"publisher","DOI":"10.1038\/nrm2774"},{"key":"2023041304134991000_34.44.14717.29","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2010.09.039"},{"key":"2023041304134991000_34.44.14717.30","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2013.10.050"},{"key":"2023041304134991000_34.44.14717.31","doi-asserted-by":"publisher","DOI":"10.1111\/j.1471-4159.1991.tb02061.x"},{"key":"2023041304134991000_34.44.14717.32","doi-asserted-by":"crossref","first-page":"6374","DOI":"10.1523\/JNEUROSCI.20-17-06374.2000","article-title":"KIF5C, a novel neuronal kinesin enriched in motor neurons","volume":"20","author":"Kanai","year":"2000","journal-title":"J Neurosci"},{"key":"2023041304134991000_34.44.14717.33","doi-asserted-by":"publisher","DOI":"10.1007\/s10048-012-0324-y"},{"key":"2023041304134991000_34.44.14717.34","doi-asserted-by":"publisher","DOI":"10.1177\/1073858412451655"},{"key":"2023041304134991000_34.44.14717.35","doi-asserted-by":"publisher","DOI":"10.1038\/nature11992"},{"key":"2023041304134991000_34.44.14717.36","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.21343"},{"key":"2023041304134991000_34.44.14717.37","doi-asserted-by":"publisher","DOI":"10.1093\/bioinformatics\/btm404"},{"key":"2023041304134991000_34.44.14717.38","doi-asserted-by":"publisher","DOI":"10.1242\/dev.012377"},{"key":"2023041304134991000_34.44.14717.39","doi-asserted-by":"publisher","DOI":"10.1038\/ng.376"},{"key":"2023041304134991000_34.44.14717.40","doi-asserted-by":"publisher","DOI":"10.1093\/nar\/gkt376"},{"key":"2023041304134991000_34.44.14717.41","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.0223-06.2006"},{"key":"2023041304134991000_34.44.14717.42","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.111145398"},{"key":"2023041304134991000_34.44.14717.43","doi-asserted-by":"publisher","DOI":"10.1242\/dev.048645"},{"key":"2023041304134991000_34.44.14717.44","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2012.10.012"},{"key":"2023041304134991000_34.44.14717.45","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(94)90012-4"},{"key":"2023041304134991000_34.44.14717.46","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-313X.1998.00124.x"},{"key":"2023041304134991000_34.44.14717.47","doi-asserted-by":"publisher","DOI":"10.1242\/dev.046425"},{"key":"2023041304134991000_34.44.14717.48","doi-asserted-by":"publisher","DOI":"10.1002\/dneu.22049"},{"key":"2023041304134991000_34.44.14717.49","doi-asserted-by":"publisher","DOI":"10.1172\/JCI37537"},{"key":"2023041304134991000_34.44.14717.50","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.90.19.9181"},{"key":"2023041304134991000_34.44.14717.51","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1327-12.2012"},{"key":"2023041304134991000_34.44.14717.52","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2613"},{"key":"2023041304134991000_34.44.14717.53","doi-asserted-by":"publisher","DOI":"10.1016\/j.febslet.2013.09.049"},{"key":"2023041304134991000_34.44.14717.54","doi-asserted-by":"publisher","DOI":"10.1086\/344210"},{"key":"2023041304134991000_34.44.14717.55","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0808953105"},{"key":"2023041304134991000_34.44.14717.56","doi-asserted-by":"publisher","DOI":"10.1002\/gene.20196"},{"key":"2023041304134991000_34.44.14717.57","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(91)90264-Y"},{"key":"2023041304134991000_34.44.14717.58","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2007.137596"},{"key":"2023041304134991000_34.44.14717.59","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a011304"},{"key":"2023041304134991000_34.44.14717.60","doi-asserted-by":"publisher","DOI":"10.1038\/nrg3141"},{"key":"2023041304134991000_34.44.14717.61","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M600522200"},{"key":"2023041304134991000_34.44.14717.62","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(00)81459-2"},{"key":"2023041304134991000_34.44.14717.63","doi-asserted-by":"publisher","DOI":"10.1097\/WCO.0b013e328364c04b"},{"key":"2023041304134991000_34.44.14717.64","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2012.11.027"},{"key":"2023041304134991000_34.44.14717.65","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.21354"},{"key":"2023041304134991000_34.44.14717.66","doi-asserted-by":"publisher","DOI":"10.1016\/S0076-6879(00)28419-X"},{"key":"2023041304134991000_34.44.14717.67","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201108111"},{"key":"2023041304134991000_34.44.14717.68","doi-asserted-by":"publisher","DOI":"10.1093\/bioinformatics\/btp033"},{"key":"2023041304134991000_34.44.14717.69","unstructured":"Westerfield M (1995) The zebrafish book: a guide for the laboratory use of Zebrafish (Danio rerio) (University of Oregon, Eugene, OR), Ed 3."},{"key":"2023041304134991000_34.44.14717.70","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2121-6-35"},{"key":"2023041304134991000_34.44.14717.71","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200301026"},{"key":"2023041304134991000_34.44.14717.72","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(01)00363-4"}],"container-title":["The Journal of Neuroscience"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1523\/JNEUROSCI.2770-14.2014","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,4,13]],"date-time":"2023-04-13T22:16:14Z","timestamp":1681424174000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.jneurosci.org\/lookup\/doi\/10.1523\/JNEUROSCI.2770-14.2014"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,10,29]]},"references-count":72,"journal-issue":{"issue":"44","published-online":{"date-parts":[[2014,10,29]]},"published-print":{"date-parts":[[2014,10,29]]}},"alternative-id":["10.1523\/JNEUROSCI.2770-14.2014"],"URL":"https:\/\/doi.org\/10.1523\/jneurosci.2770-14.2014","relation":{},"ISSN":["0270-6474","1529-2401"],"issn-type":[{"value":"0270-6474","type":"print"},{"value":"1529-2401","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,10,29]]}}}