{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,13]],"date-time":"2026-06-13T01:56:31Z","timestamp":1781315791970,"version":"3.54.1"},"reference-count":43,"publisher":"Public Library of Science (PLoS)","issue":"7","license":[{"start":{"date-parts":[[2017,7,5]],"date-time":"2017-07-05T00:00:00Z","timestamp":1499212800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100006545","name":"National Institute on Minority Health and Health Disparities","doi-asserted-by":"publisher","award":["2G12MD007595-06"],"award-info":[{"award-number":["2G12MD007595-06"]}],"id":[{"id":"10.13039\/100006545","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"publisher","award":["8P20GM103424"],"award-info":[{"award-number":["8P20GM103424"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"publisher","award":["R01GM066328"],"award-info":[{"award-number":["R01GM066328"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"publisher","award":["5UL1GM118967"],"award-info":[{"award-number":["5UL1GM118967"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"DOI":"10.1371\/journal.pone.0180353","type":"journal-article","created":{"date-parts":[[2017,7,5]],"date-time":"2017-07-05T17:48:04Z","timestamp":1499276884000},"page":"e0180353","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":31,"title":["Characterization of kinesin switch I mutations that cause hereditary spastic paraplegia"],"prefix":"10.1371","volume":"12","author":[{"given":"Scott","family":"Jennings","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Madeline","family":"Chenevert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liqiong","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Madhusoodanan","family":"Mottamal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward J.","family":"Wojcik","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6711-9175","authenticated-orcid":true,"given":"Thomas M.","family":"Huckaba","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2017,7,5]]},"reference":[{"issue":"1","key":"ref1","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1136\/jnnp.37.1.8","article-title":"Strumpell's familial spastic paraplegia: genetics and neuropathology","volume":"37","author":"WM Behan","year":"1974","journal-title":"J Neurol Neurosurg Psychiatry"},{"issue":"1\u20132","key":"ref2","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.jns.2012.03.025","article-title":"Hereditary spastic paraplegias with autosomal dominant, recessive, X-linked, or maternal trait of inheritance","volume":"318","author":"J Finsterer","year":"2012","journal-title":"J Neurol Sci"},{"issue":"Suppl","key":"ref3","doi-asserted-by":"crossref","first-page":"S16","DOI":"10.1016\/0303-8467(92)90010-Z","article-title":"The neuropathology of hereditary spastic paraparesis","volume":"94","author":"RP Bruyn","year":"1992","journal-title":"Clin Neurol Neurosurg"},{"issue":"6\u20137","key":"ref4","doi-asserted-by":"crossref","first-page":"505","DOI":"10.1016\/j.neurol.2015.02.017","article-title":"Clinical and genetic heterogeneity in hereditary spastic paraplegias: from SPG1 to SPG72 and still counting","volume":"171","author":"S Klebe","year":"2015","journal-title":"Rev Neurol (Paris)"},{"issue":"2","key":"ref5","doi-asserted-by":"crossref","first-page":"150","DOI":"10.1136\/jnnp.69.2.150","article-title":"Hereditary spastic paraparesis: a review of new developments","volume":"69","author":"C McDermott","year":"2000","journal-title":"J Neurol Neurosurg Psychiatry"},{"issue":"2","key":"ref6","doi-asserted-by":"crossref","first-page":"E376","DOI":"10.1002\/humu.20920","article-title":"Complicated forms of autosomal dominant hereditary spastic paraplegia are frequent in SPG10","volume":"30","author":"C Goizet","year":"2009","journal-title":"Hum Mutat"},{"issue":"1","key":"ref7","doi-asserted-by":"crossref","first-page":"47","DOI":"10.1007\/s10048-005-0027-8","article-title":"Mutation in KIF5A can also cause adult-onset hereditary spastic paraplegia","volume":"7","author":"MA Blair","year":"2006","journal-title":"Neurogenetics"},{"issue":"6","key":"ref8","doi-asserted-by":"crossref","first-page":"702","DOI":"10.1136\/jnnp-2014-308625","article-title":"Hereditary spastic paraplegia: a novel mutation and expansion of the phenotype variability in SPG10","volume":"86","author":"L Carosi","year":"2015","journal-title":"J Neurol Neurosurg Psychiatry"},{"issue":"2","key":"ref9","doi-asserted-by":"crossref","first-page":"157","DOI":"10.1111\/j.1399-0004.2011.01717.x","article-title":"Mutations in the motor and stalk domains of KIF5A in spastic paraplegia type 10 and in axonal Charcot-Marie-Tooth type 2","volume":"82","author":"C Crimella","year":"2011","journal-title":"Clin Genet"},{"issue":"6","key":"ref10","doi-asserted-by":"crossref","first-page":"1108","DOI":"10.1212\/01.WNL.0000138731.60693.D2","article-title":"Evidence of kinesin heavy chain (KIF5A) involvement in pure hereditary spastic paraplegia","volume":"63","author":"M Fichera","year":"2004","journal-title":"Neurology"},{"issue":"7","key":"ref11","doi-asserted-by":"crossref","first-page":"612","DOI":"10.1212\/WNL.0000000000000691","article-title":"Extended phenotypic spectrum of KIF5A mutations: From spastic paraplegia to axonal neuropathy","volume":"83","author":"YT Liu","year":"2014","journal-title":"Neurology"},{"issue":"2","key":"ref12","doi-asserted-by":"crossref","first-page":"284","DOI":"10.1001\/archneur.63.2.284","article-title":"A missense mutation in the coiled-coil domain of the KIF5A gene and late-onset hereditary spastic paraplegia","volume":"63","author":"M Lo Giudice","year":"2006","journal-title":"Arch Neurol"},{"issue":"1\u20132","key":"ref13","doi-asserted-by":"crossref","first-page":"422","DOI":"10.1016\/j.jns.2015.08.1529","article-title":"Identification of two novel KIF5A mutations in hereditary spastic paraplegia associated with mild peripheral neuropathy","volume":"358","author":"E Lopez","year":"2015","journal-title":"J Neurol Sci"},{"issue":"1\u20132","key":"ref14","doi-asserted-by":"crossref","first-page":"218","DOI":"10.1016\/j.jns.2014.05.063","article-title":"A novel KIF5A mutation in an Italian family marked by spastic paraparesis and congenital deafness","volume":"343","author":"M Muglia","year":"2014","journal-title":"J Neurol Sci"},{"issue":"4","key":"ref15","doi-asserted-by":"crossref","first-page":"665","DOI":"10.1007\/s10072-010-0445-8","article-title":"A novel mutation in KIF5A gene causing hereditary spastic paraplegia with axonal neuropathy","volume":"32","author":"O Musumeci","year":"2011","journal-title":"Neurol Sci"},{"issue":"5","key":"ref16","doi-asserted-by":"crossref","first-page":"1189","DOI":"10.1086\/344210","article-title":"A kinesin heavy chain (KIF5A) mutation in hereditary spastic paraplegia (SPG10)","volume":"71","author":"E Reid","year":"2002","journal-title":"Am J Hum Genet"},{"issue":"3","key":"ref17","doi-asserted-by":"crossref","first-page":"153","DOI":"10.1097\/CND.0000000000000063","article-title":"A novel mutation in motor domain of KIF5A associated with an HSP\/axonal neuropathy phenotype","volume":"16","author":"F Rinaldi","year":"2015","journal-title":"J Clin Neuromuscul Dis"},{"issue":"5","key":"ref18","doi-asserted-by":"crossref","first-page":"584","DOI":"10.1136\/jnnp.2007.137596","article-title":"SPG10 is a rare cause of spastic paraplegia in European families","volume":"79","author":"R Schule","year":"2008","journal-title":"J Neurol Neurosurg Psychiatry"},{"issue":"7","key":"ref19","doi-asserted-by":"crossref","first-page":"1090","DOI":"10.1007\/s00415-008-0840-8","article-title":"A novel KIF5A\/SPG10 mutation in spastic paraplegia associated with axonal neuropathy","volume":"255","author":"A Tessa","year":"2008","journal-title":"J Neurol"},{"issue":"1","key":"ref20","doi-asserted-by":"crossref","first-page":"59","DOI":"10.1016\/j.ceb.2008.12.002","article-title":"Walking the walk: how kinesin and dynein coordinate their steps","volume":"21","author":"A Gennerich","year":"2009","journal-title":"Curr Opin Cell Biol"},{"issue":"2","key":"ref21","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1083\/jcb.135.2.291","article-title":"Switches, latches, and amplifiers: common themes of G proteins and molecular motors","volume":"135","author":"RD Vale","year":"1996","journal-title":"J Cell Biol"},{"issue":"6574","key":"ref22","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1038\/380550a0","article-title":"Crystal structure of the kinesin motor domain reveals a structural similarity to myosin","volume":"380","author":"FJ Kull","year":"1996","journal-title":"Nature"},{"issue":"8","key":"ref23","doi-asserted-by":"crossref","first-page":"5859","DOI":"10.1074\/jbc.M109.071233","article-title":"ATP hydrolysis in Eg5 kinesin involves a catalytic two-water mechanism","volume":"285","author":"CL Parke","year":"2010","journal-title":"J Biol Chem"},{"issue":"11","key":"ref24","doi-asserted-by":"crossref","first-page":"2611","DOI":"10.1093\/emboj\/20.11.2611","article-title":"A structural pathway for activation of the kinesin motor ATPase","volume":"20","author":"M Yun","year":"2001","journal-title":"EMBO J"},{"issue":"1","key":"ref25","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1046\/j.1432-1327.1999.00341.x","article-title":"Motor proteins of the kinesin family. Structures, variations, and nucleotide binding sites","volume":"262","author":"S Sack","year":"1999","journal-title":"Eur J Biochem"},{"key":"ref26","doi-asserted-by":"crossref","first-page":"5364","DOI":"10.1038\/ncomms6364","article-title":"The structure of apo-kinesin bound to tubulin links the nucleotide cycle to movement","volume":"5","author":"L Cao","year":"2014","journal-title":"Nat Commun"},{"issue":"8","key":"ref27","doi-asserted-by":"crossref","first-page":"1001","DOI":"10.1038\/nsmb.2624","article-title":"Structure of a kinesin-tubulin complex and implications for kinesin motility","volume":"20","author":"B Gigant","year":"2013","journal-title":"Nat Struct Mol Biol"},{"issue":"2","key":"ref28","doi-asserted-by":"crossref","first-page":"207","DOI":"10.1016\/S0092-8674(00)80329-3","article-title":"Microtubule interaction site of the kinesin motor","volume":"90","author":"G Woehlke","year":"1997","journal-title":"Cell"},{"issue":"9","key":"ref29","doi-asserted-by":"crossref","first-page":"7457","DOI":"10.1074\/jbc.M110.167023","article-title":"Kinesin-73 is a processive motor that localizes to Rab5-containing organelles","volume":"286","author":"TM Huckaba","year":"2011","journal-title":"J Biol Chem"},{"issue":"1","key":"ref30","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1016\/S1046-5928(03)00218-3","article-title":"Purification of brain tubulin through two cycles of polymerization-depolymerization in a high-molarity buffer","volume":"32","author":"M Castoldi","year":"2003","journal-title":"Protein Expr Purif"},{"issue":"1","key":"ref31","doi-asserted-by":"crossref","first-page":"283","DOI":"10.1006\/jcph.1999.6201","article-title":"NAMD2: Greater scalability for parallel molecular dynamics","volume":"151","author":"L Kale","year":"1999","journal-title":"Journal of Computational Physics"},{"issue":"18","key":"ref32","doi-asserted-by":"crossref","first-page":"3586","DOI":"10.1021\/jp973084f","article-title":"All-atom empirical potential for molecular modeling and dynamics studies of proteins","volume":"102","author":"AD MacKerell","year":"1998","journal-title":"J Phys Chem B"},{"issue":"17","key":"ref33","doi-asserted-by":"crossref","first-page":"6314","DOI":"10.1073\/pnas.85.17.6314","article-title":"Kinesin ATPase: rate-limiting ADP release","volume":"85","author":"DD Hackney","year":"1988","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"6516","key":"ref34","doi-asserted-by":"crossref","first-page":"671","DOI":"10.1038\/373671a0","article-title":"Pathway of processive ATP hydrolysis by kinesin","volume":"373","author":"SP Gilbert","year":"1995","journal-title":"Nature"},{"issue":"40","key":"ref35","doi-asserted-by":"crossref","first-page":"13233","DOI":"10.1021\/bi00040a039","article-title":"Kinetic mechanism of kinesin motor domain","volume":"34","author":"YZ Ma","year":"1995","journal-title":"Biochemistry"},{"issue":"Pt 1","key":"ref36","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1242\/jcs.115.1.15","article-title":"Kinesin: switch I & II and the motor mechanism","volume":"115","author":"FJ Kull","year":"2002","journal-title":"J Cell Sci"},{"issue":"2","key":"ref37","doi-asserted-by":"crossref","first-page":"148","DOI":"10.1016\/j.ymeth.2009.07.005","article-title":"Identification of catalytic metal ion ligands in ribozymes","volume":"49","author":"JK Frederiksen","year":"2009","journal-title":"Methods"},{"issue":"1","key":"ref38","doi-asserted-by":"crossref","first-page":"122","DOI":"10.1038\/nsmb.2190","article-title":"A metal switch for controlling the activity of molecular motor proteins","volume":"19","author":"JC Cochran","year":"2012","journal-title":"Nat Struct Mol Biol"},{"issue":"16","key":"ref39","doi-asserted-by":"crossref","first-page":"11352","DOI":"10.1016\/S0021-9258(19)49917-1","article-title":"A kinetic study of the kinesin ATPase","volume":"267","author":"A Sadhu","year":"1992","journal-title":"J Biol Chem"},{"issue":"9","key":"ref40","doi-asserted-by":"crossref","first-page":"1245","DOI":"10.1093\/hmg\/ddn014","article-title":"Effect of spastic paraplegia mutations in KIF5A kinesin on transport activity","volume":"17","author":"B Ebbing","year":"2008","journal-title":"Hum Mol Genet"},{"issue":"1","key":"ref41","doi-asserted-by":"crossref","first-page":"173","DOI":"10.1534\/genetics.112.140798","article-title":"Three routes to suppression of the neurodegenerative phenotypes caused by kinesin heavy chain mutations","volume":"192","author":"I Djagaeva","year":"2012","journal-title":"Genetics"},{"issue":"11","key":"ref42","doi-asserted-by":"crossref","first-page":"e1003066","DOI":"10.1371\/journal.pgen.1003066","article-title":"Spastic paraplegia mutation N256S in the neuronal microtubule motor KIF5A disrupts axonal transport in a Drosophila HSP model","volume":"8","author":"P Fuger","year":"2012","journal-title":"PLoS Genet"},{"key":"ref43","doi-asserted-by":"crossref","first-page":"42558","DOI":"10.1038\/srep42558","article-title":"The structural switch of nucleotide-free kinesin","volume":"7","author":"L Cao","year":"2017","journal-title":"Sci Rep"}],"container-title":["PLOS ONE"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/dx.plos.org\/10.1371\/journal.pone.0180353","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,7,30]],"date-time":"2022-07-30T13:41:52Z","timestamp":1659188512000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0180353"}},"subtitle":[],"editor":[{"given":"Alexander F.","family":"Palazzo","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2017,7,5]]},"references-count":43,"journal-issue":{"issue":"7","published-online":{"date-parts":[[2017,7,5]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pone.0180353","relation":{},"ISSN":["1932-6203"],"issn-type":[{"value":"1932-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2017,7,5]]}}}