{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,11]],"date-time":"2026-04-11T21:57:41Z","timestamp":1775944661913,"version":"3.50.1"},"reference-count":58,"publisher":"The Company of Biologists","issue":"19","license":[{"start":{"date-parts":[[2025,10,1]],"date-time":"2025-10-01T00:00:00Z","timestamp":1759276800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-2\/"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82370647"],"award-info":[{"award-number":["82370647"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82170642"],"award-info":[{"award-number":["82170642"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["32430026"],"award-info":[{"award-number":["32430026"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["32270773"],"award-info":[{"award-number":["32270773"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["32470730"],"award-info":[{"award-number":["32470730"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["32270721"],"award-info":[{"award-number":["32270721"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["32021002"],"award-info":[{"award-number":["32021002"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["92254306"],"award-info":[{"award-number":["92254306"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100012166","name":"National Key R&D Program of China","doi-asserted-by":"crossref","award":["2022YFA1302700"],"award-info":[{"award-number":["2022YFA1302700"]}],"id":[{"id":"10.13039\/501100012166","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100012166","name":"National Key R&D Program of China","doi-asserted-by":"crossref","award":["2024YFA1307301"],"award-info":[{"award-number":["2024YFA1307301"]}],"id":[{"id":"10.13039\/501100012166","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100004147","name":"Tsinghua University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100004147","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,10,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>KIF1A-associated neurological disorder (KAND) arises from mutations in the microtubule motor KIF1A, disrupting synaptic vesicle transport. Here, we investigate the pathogenic T99M substitution in the P-loop of KIF1A, which induces steric hindrance, impairing ATP\/ADP coordination and motor activity. Using CRISPR-engineered Caenorhabditis elegans expressing the homologous UNC-104(T95M) mutation, we conducted forward genetic screens and identified recurrent intragenic suppressors (T95V and T95I; T95V\/I) that restored animal motility and synaptic vesicle distribution. Molecular dynamics simulations revealed that replacing the methionine residue with valine or isoleucine residues alleviated steric clashes in the nucleotide-binding pocket and stabilized Mg2+-ATP coordination. Biochemical assays showed that T95V\/I partially recovered microtubule gliding velocity and processivity, demonstrating that even modest motor reactivation mitigates neuronal dysfunction. Inspired by prior success showing that treatment with the plant flavonol fisetin rescues the effects of the KIF1A R11Q variant, we propose allele-specific conformational stabilization as a therapeutic strategy for KAND. Our findings highlight the structural plasticity of motor domains and provide a framework for precision therapies targeting pathogenic variants through genetic suppressors.<\/jats:p>","DOI":"10.1242\/jcs.264216","type":"journal-article","created":{"date-parts":[[2025,9,18]],"date-time":"2025-09-18T09:23:18Z","timestamp":1758187398000},"source":"Crossref","is-referenced-by-count":1,"title":["Allele-specific conformational rescue of KIF1A T99M by genetic suppressors in a <i>C. elegans<\/i> model of KIF1A-associated neurological disorder"],"prefix":"10.1242","volume":"138","author":[{"given":"Zihan","family":"Chen","sequence":"first","affiliation":[{"name":"School of Basic Medicine, Tongji Medical College, Huazhong University of Science and Technology 1 Department of Medical Genetics , , Wuhan 430030 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yongping","family":"Chai","sequence":"additional","affiliation":[{"name":"Tsinghua-Peking Center for Life Sciences, Beijing Frontier Research Center for Biological Structure, McGovern Institute for Brain Research, State Key Laboratory of Membrane Biology, School of Life Sciences and MOE Key Laboratory for Protein Science, Tsinghua University 2 , Beijing 100084 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zhengyang","family":"Guo","sequence":"additional","affiliation":[{"name":"Tsinghua-Peking Center for Life Sciences, Beijing Frontier Research Center for Biological Structure, McGovern Institute for Brain Research, State Key Laboratory of Membrane Biology, School of Life Sciences and MOE Key Laboratory for Protein Science, Tsinghua University 2 , Beijing 100084 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xuanyu","family":"Fu","sequence":"additional","affiliation":[{"name":"Tsinghua-Peking Center for Life Sciences, Beijing Frontier Research Center for Biological Structure, McGovern Institute for Brain Research, State Key Laboratory of Membrane Biology, School of Life Sciences and MOE Key Laboratory for Protein Science, Tsinghua University 2 , Beijing 100084 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wei","family":"Li","sequence":"additional","affiliation":[{"name":"School of Medicine, Tsinghua University 3 , Beijing 100084 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jinxiang","family":"Zhang","sequence":"additional","affiliation":[{"name":"Union Hospital, Tongji Medical College, Huazhong University of Science and Technology 4 Department of Emergency Surgery , , 1277 Jiefang Avenue, Wuhan 430022 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1512-7824","authenticated-orcid":false,"given":"Guangshuo","family":"Ou","sequence":"additional","affiliation":[{"name":"Tsinghua-Peking Center for Life Sciences, Beijing Frontier Research Center for Biological Structure, McGovern Institute for Brain Research, State Key Laboratory of Membrane Biology, School of Life Sciences and MOE Key Laboratory for Protein Science, Tsinghua University 2 , Beijing 100084 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hui","family":"Wang","sequence":"additional","affiliation":[{"name":"School of Basic Medicine, Tongji Medical College, Huazhong University of Science and Technology 1 Department of Medical Genetics , , Wuhan 430030 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2025,10,14]]},"reference":[{"key":"2025101404121494400_JCS264216C1","doi-asserted-by":"publisher","first-page":"e2113795119","DOI":"10.1073\/pnas.2113795119","article-title":"De novo mutations in KIF1A-associated neuronal disorder (KAND) dominant-negatively inhibit motor activity and axonal transport of synaptic vesicle precursors","volume":"119","author":"Anazawa","year":"2022","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2025101404121494400_JCS264216C2","doi-asserted-by":"publisher","first-page":"5530","DOI":"10.1038\/s41467-024-48720-4","article-title":"Cryo-EM unveils kinesin KIF1A's processivity mechanism and the impact of its pathogenic variant P305L","volume":"15","author":"Benoit","year":"2024","journal-title":"Nat. Commun."},{"key":"2025101404121494400_JCS264216C3","doi-asserted-by":"publisher","first-page":"100026","DOI":"10.1016\/j.xhgg.2021.100026","article-title":"Genotype and defects in microtubule-based motility correlate with clinical severity in KIF1A-associated neurological disorder","volume":"2","author":"Boyle","year":"2021","journal-title":"HGG Adv."},{"key":"2025101404121494400_JCS264216C4","doi-asserted-by":"publisher","first-page":"71","DOI":"10.1093\/genetics\/77.1.71","article-title":"The genetics of Caenorhabditis elegans","volume":"77","author":"Brenner","year":"1974","journal-title":"Genetics"},{"key":"2025101404121494400_JCS264216C5","doi-asserted-by":"publisher","first-page":"e202004227","DOI":"10.1083\/jcb.202004227","article-title":"Pathogenic mutations in the kinesin-3 motor KIF1A diminish force generation and movement through allosteric mechanisms","volume":"220","author":"Budaitis","year":"2021","journal-title":"J. Cell Biol."},{"key":"2025101404121494400_JCS264216C6","doi-asserted-by":"publisher","first-page":"787","DOI":"10.1016\/S0896-6273(01)00532-3","article-title":"UNC-16, a JNK-signaling scaffold protein, regulates vesicle transport in C. elegans","volume":"32","author":"Byrd","year":"2001","journal-title":"Neuron"},{"key":"2025101404121494400_JCS264216C7","doi-asserted-by":"publisher","first-page":"699","DOI":"10.1038\/s41580-022-00491-w","article-title":"Selective motor activation in organelle transport along axons","volume":"23","author":"Cason","year":"2022","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2025101404121494400_JCS264216C8","doi-asserted-by":"publisher","first-page":"e2311936121","DOI":"10.1073\/pnas.2311936121","article-title":"A plant flavonol and genetic suppressors rescue a pathogenic mutation associated with kinesin in neurons","volume":"121","author":"Chai","year":"2024","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2025101404121494400_JCS264216C9","doi-asserted-by":"publisher","first-page":"18429","DOI":"10.1073\/pnas.1905690116","article-title":"Disease-associated mutations hyperactivate KIF1A motility and anterograde axonal transport of synaptic vesicle precursors","volume":"116","author":"Chiba","year":"2019","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2025101404121494400_JCS264216C10","doi-asserted-by":"publisher","first-page":"jcs260742","DOI":"10.1242\/jcs.260742","article-title":"Insight into the regulation of axonal transport from the study of KIF1A-associated neurological disorder","volume":"136","author":"Chiba","year":"2023","journal-title":"J. Cell Sci."},{"key":"2025101404121494400_JCS264216C11","doi-asserted-by":"publisher","first-page":"2684","DOI":"10.1007\/s00415-015-7899-9","article-title":"Variants in KIF1A gene in dominant and sporadic forms of hereditary spastic paraparesis","volume":"262","author":"Citterio","year":"2015","journal-title":"J. Neurol."},{"key":"2025101404121494400_JCS264216C12","doi-asserted-by":"publisher","first-page":"e1009940","DOI":"10.1371\/journal.pgen.1009940","article-title":"Motor domain-mediated autoinhibition dictates axonal transport by the kinesin UNC-104\/KIF1A","volume":"17","author":"Cong","year":"2021","journal-title":"PLoS Genet."},{"key":"2025101404121494400_JCS264216C13","doi-asserted-by":"publisher","first-page":"1028","DOI":"10.1038\/nmeth.2641","article-title":"Engineering the Caenorhabditis elegans genome using Cas9-triggered homologous recombination","volume":"10","author":"Dickinson","year":"2013","journal-title":"Nat. Methods"},{"key":"2025101404121494400_JCS264216C58","doi-asserted-by":"publisher","first-page":"e1005659","DOI":"10.1371\/journal.pcbi.1005659","article-title":"OpenMM 7: Rapid development of high performance algorithms for molecular dynamics","volume":"13","author":"Eastman","year":"2017","journal-title":"PLoS Comput. Biol."},{"key":"2025101404121494400_JCS264216C14","doi-asserted-by":"publisher","first-page":"623","DOI":"10.1002\/acn3.198","article-title":"De novo mutations in KIF1A cause progressive encephalopathy and brain atrophy","volume":"2","author":"Esmaeeli Nieh","year":"2015","journal-title":"Ann. Clin. Transl. Neurol."},{"key":"2025101404121494400_JCS264216C15","doi-asserted-by":"publisher","first-page":"3838","DOI":"10.1242\/dev.098723","article-title":"Developmental stage-dependent transcriptional regulatory pathways control neuroblast lineage progression","volume":"140","author":"Feng","year":"2013","journal-title":"Development"},{"key":"2025101404121494400_JCS264216C16","doi-asserted-by":"publisher","first-page":"419","DOI":"10.3389\/fncel.2019.00419","article-title":"Going too far is the same as falling short(dagger): kinesin-3 family members in hereditary spastic paraplegia","volume":"13","author":"Gabrych","year":"2019","journal-title":"Front. Cell Neurosci."},{"key":"2025101404121494400_JCS264216C17","doi-asserted-by":"publisher","first-page":"221","DOI":"10.1016\/S0091-679X(10)95013-9","article-title":"Microtubule dynamics reconstituted in vitro and imaged by single-molecule fluorescence microscopy","volume":"95","author":"Gell","year":"2010","journal-title":"Methods Cell Biol."},{"key":"2025101404121494400_JCS264216C18","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1007\/978-1-61779-252-6_2","article-title":"Purification of tubulin from porcine brain","volume":"777","author":"Gell","year":"2011","journal-title":"Methods Mol. Biol."},{"key":"2025101404121494400_JCS264216C19","doi-asserted-by":"publisher","first-page":"306","DOI":"10.1016\/j.ajhg.2011.02.001","article-title":"Excess of de novo deleterious mutations in genes associated with glutamatergic systems in nonsyndromic intellectual disability","volume":"88","author":"Hamdan","year":"2011","journal-title":"Am. J. Hum. Genet."},{"key":"2025101404121494400_JCS264216C20","doi-asserted-by":"publisher","first-page":"1089","DOI":"10.1152\/physrev.00023.2007","article-title":"Intracellular transport and kinesin superfamily proteins, KIFs: structure, function, and dynamics","volume":"88","author":"Hirokawa","year":"2008","journal-title":"Physiol. Rev."},{"key":"2025101404121494400_JCS264216C21","doi-asserted-by":"publisher","first-page":"877","DOI":"10.1038\/nrm2807","article-title":"The mechanisms of kinesin motor motility: lessons from the monomeric motor KIF1A","volume":"10","author":"Hirokawa","year":"2009","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2025101404121494400_JCS264216C22","doi-asserted-by":"publisher","first-page":"1114","DOI":"10.1177\/0883073816639718","article-title":"Novel de novo mutations in KIF1A as a cause of hereditary spastic paraplegia with progressive central nervous system involvement","volume":"31","author":"Hotchkiss","year":"2016","journal-title":"J. Child Neurol."},{"key":"2025101404121494400_JCS264216C23","doi-asserted-by":"publisher","first-page":"71","DOI":"10.1038\/nmeth.4067","article-title":"CHARMM36m: an improved force field for folded and intrinsically disordered proteins","volume":"14","author":"Huang","year":"2017","journal-title":"Nat. Methods"},{"key":"2025101404121494400_JCS264216C24","doi-asserted-by":"publisher","first-page":"e28948","DOI":"10.7554\/eLife.28948","article-title":"Kinesin motility is driven by subdomain dynamics","volume":"6","author":"Hwang","year":"2017","journal-title":"eLife"},{"key":"2025101404121494400_JCS264216C25","doi-asserted-by":"publisher","first-page":"478","DOI":"10.1016\/0076-6879(91)96041-O","article-title":"Preparation of modified tubulins","volume":"196","author":"Hyman","year":"1991","journal-title":"Methods Enzymol."},{"key":"2025101404121494400_JCS264216C26","doi-asserted-by":"publisher","first-page":"1859","DOI":"10.1002\/jcc.20945","article-title":"CHARMM-GUI: a web-based graphical user interface for CHARMM","volume":"29","author":"Jo","year":"2008","journal-title":"J. Comput. Chem."},{"key":"2025101404121494400_JCS264216C27","doi-asserted-by":"publisher","first-page":"439","DOI":"10.1038\/35078000","article-title":"Switch-based mechanism of kinesin motors","volume":"411","author":"Kikkawa","year":"2001","journal-title":"Nature"},{"key":"2025101404121494400_JCS264216C28","doi-asserted-by":"publisher","first-page":"1879","DOI":"10.1002\/jcc.24829","article-title":"CHARMM-GUI ligand reader and modeler for CHARMM force field generation of small molecules","volume":"38","author":"Kim","year":"2017","journal-title":"J. Comput. Chem."},{"key":"2025101404121494400_JCS264216C29","doi-asserted-by":"publisher","first-page":"645","DOI":"10.1038\/ejhg.2011.261","article-title":"KIF1A missense mutations in SPG30, an autosomal recessive spastic paraplegia: distinct phenotypes according to the nature of the mutations","volume":"20","author":"Klebe","year":"2012","journal-title":"Eur. J. Hum. Genet."},{"key":"2025101404121494400_JCS264216C30","doi-asserted-by":"publisher","first-page":"e1001200","DOI":"10.1371\/journal.pgen.1001200","article-title":"The Caenorhabditis elegans Kinesin-3 motor UNC-104\/KIF1A is degraded upon loss of specific binding to cargo","volume":"6","author":"Kumar","year":"2010","journal-title":"PLoS Genet."},{"key":"2025101404121494400_JCS264216C31","doi-asserted-by":"publisher","first-page":"949","DOI":"10.1038\/ejhg.2015.217","article-title":"De novo dominant variants affecting the motor domain of KIF1A are a cause of PEHO syndrome","volume":"24","author":"Langlois","year":"2016","journal-title":"Eur. J. Hum. Genet."},{"key":"2025101404121494400_JCS264216C32","doi-asserted-by":"publisher","first-page":"69","DOI":"10.1002\/humu.22709","article-title":"De novo mutations in the motor domain of KIF1A cause cognitive impairment, spastic paraparesis, axonal neuropathy, and cerebellar atrophy","volume":"36","author":"Lee","year":"2015","journal-title":"Hum. Mutat."},{"key":"2025101404121494400_JCS264216C33","doi-asserted-by":"publisher","first-page":"605","DOI":"10.1016\/j.cub.2015.12.068","article-title":"The neuronal kinesin UNC-104\/KIF1A is a key regulator of synaptic aging and insulin signaling-regulated memory","volume":"26","author":"Li","year":"2016","journal-title":"Curr. Biol."},{"key":"2025101404121494400_JCS264216C34","doi-asserted-by":"publisher","first-page":"273","DOI":"10.1111\/tra.12142","article-title":"In vivo neuron-wide analysis of synaptic vesicle precursor trafficking","volume":"15","author":"Maeder","year":"2014","journal-title":"Traffic"},{"key":"2025101404121494400_JCS264216C35","doi-asserted-by":"publisher","first-page":"147","DOI":"10.3390\/ph16020147","article-title":"KIF1A-associated neurological disorder: an overview of a rare mutational disease","volume":"16","author":"Nair","year":"2023","journal-title":"Pharmaceuticals (Basel)"},{"key":"2025101404121494400_JCS264216C36","doi-asserted-by":"publisher","first-page":"117","DOI":"10.1111\/jns.12368","article-title":"KIF1A-related disorders in children: a wide spectrum of central and peripheral nervous system involvement","volume":"25","author":"Nemani","year":"2020","journal-title":"J. Peripher. Nerv. Syst."},{"key":"2025101404121494400_JCS264216C37","doi-asserted-by":"publisher","first-page":"1269","DOI":"10.1038\/ncb1785","article-title":"KIF1Bbeta- and KIF1A-mediated axonal transport of presynaptic regulator Rab3 occurs in a GTP-dependent manner through DENN\/MADD","volume":"10","author":"Niwa","year":"2008","journal-title":"Nat. Cell Biol."},{"key":"2025101404121494400_JCS264216C38","doi-asserted-by":"publisher","first-page":"2129","DOI":"10.1016\/j.celrep.2016.07.043","article-title":"Autoinhibition of a neuronal kinesin UNC-104\/KIF1A regulates the size and density of synapses","volume":"16","author":"Niwa","year":"2016","journal-title":"Cell Rep."},{"key":"2025101404121494400_JCS264216C39","doi-asserted-by":"publisher","first-page":"769","DOI":"10.1016\/0092-8674(95)90538-3","article-title":"The neuron-specific kinesin superfamily protein KIF1A is a unique monomeric motor for anterograde axonal transport of synaptic vesicle precursors","volume":"81","author":"Okada","year":"1995","journal-title":"Cell"},{"key":"2025101404121494400_JCS264216C40","doi-asserted-by":"publisher","first-page":"3851","DOI":"10.1016\/j.cub.2023.07.052","article-title":"End-binding protein 1 promotes specific motor-cargo association in the cell body prior to axonal delivery of dense core vesicles","volume":"33","author":"Park","year":"2023","journal-title":"Curr. Biol."},{"key":"2025101404121494400_JCS264216C41","doi-asserted-by":"publisher","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":"Parke","year":"2010","journal-title":"J. Biol. Chem."},{"key":"2025101404121494400_JCS264216C42","doi-asserted-by":"publisher","first-page":"40","DOI":"10.1038\/s41431-019-0497-z","article-title":"KIF1A variants are a frequent cause of autosomal dominant hereditary spastic paraplegia","volume":"28","author":"Pennings","year":"2020","journal-title":"Eur. J. Hum. Genet."},{"key":"2025101404121494400_JCS264216C43","doi-asserted-by":"publisher","first-page":"20982","DOI":"10.1073\/pnas.0810359105","article-title":"Genetic analysis of crawling and swimming locomotory patterns in C. elegans","volume":"105","author":"Pierce-Shimomura","year":"2008","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2025101404121494400_JCS264216C44","doi-asserted-by":"publisher","first-page":"656","DOI":"10.3390\/biom15050656","article-title":"Distinct clinical phenotypes in KIF1A-associated neurological disorders result from different amino acid substitutions at the same residue in KIF1A","volume":"15","author":"Rao","year":"2025","journal-title":"Biomolecules"},{"key":"2025101404121494400_JCS264216C45","doi-asserted-by":"publisher","first-page":"219","DOI":"10.1016\/j.ajhg.2011.06.013","article-title":"KIF1A, an axonal transporter of synaptic vesicles, is mutated in hereditary sensory and autonomic neuropathy type 2","volume":"89","author":"Riviere","year":"2011","journal-title":"Am. J. Hum. Genet."},{"key":"2025101404121494400_JCS264216C46","doi-asserted-by":"publisher","first-page":"676","DOI":"10.1038\/nmeth.2019","article-title":"Fiji: an open-source platform for biological-image analysis","volume":"9","author":"Schindelin","year":"2012","journal-title":"Nat. Methods"},{"key":"2025101404121494400_JCS264216C47","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1016\/j.devcel.2016.06.012","article-title":"KIF1A\/UNC-104 transports ATG-9 to regulate neurodevelopment and autophagy at synapses","volume":"38","author":"Stavoe","year":"2016","journal-title":"Dev. Cell"},{"key":"2025101404121494400_JCS264216C48","doi-asserted-by":"publisher","first-page":"4329","DOI":"10.1016\/j.cell.2021.06.023","article-title":"Molecular topography of an entire nervous system","volume":"184","author":"Taylor","year":"2021","journal-title":"Cell"},{"key":"2025101404121494400_JCS264216C49","doi-asserted-by":"publisher","first-page":"1749","DOI":"10.1101\/gr.157651.113","article-title":"The million mutation project: a new approach to genetics in Caenorhabditis elegans","volume":"23","author":"Thompson","year":"2013","journal-title":"Genome Res."},{"key":"2025101404121494400_JCS264216C50","doi-asserted-by":"publisher","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":"Vale","year":"1996","journal-title":"J. Cell Biol."},{"key":"2025101404121494400_JCS264216C51","doi-asserted-by":"publisher","first-page":"4393","DOI":"10.1091\/mbc.e12-06-0444","article-title":"One-step purification of assembly-competent tubulin from diverse eukaryotic sources","volume":"23","author":"Widlund","year":"2012","journal-title":"Mol. Biol. Cell"},{"key":"2025101404121494400_JCS264216C52","doi-asserted-by":"publisher","first-page":"994","DOI":"10.1016\/j.neuron.2013.04.035","article-title":"The balance between capture and dissociation of presynaptic proteins controls the spatial distribution of synapses","volume":"78","author":"Wu","year":"2013","journal-title":"Neuron"},{"key":"2025101404121494400_JCS264216C53","doi-asserted-by":"publisher","first-page":"86","DOI":"10.1038\/s41580-024-00780-6","article-title":"Mechanism and regulation of kinesin motors","volume":"26","author":"Yildiz","year":"2025","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2025101404121494400_JCS264216C54","doi-asserted-by":"publisher","first-page":"431","DOI":"10.1083\/jcb.141.2.431","article-title":"Defect in synaptic vesicle precursor transport and neuronal cell death in KIF1A motor protein-deficient mice","volume":"141","author":"Yonekawa","year":"1998","journal-title":"J. Cell Biol."},{"key":"2025101404121494400_JCS264216C55","doi-asserted-by":"publisher","first-page":"38172","DOI":"10.1038\/srep38172","article-title":"The KIF1A homolog Unc-104 is important for spontaneous release, postsynaptic density maturation and perisynaptic scaffold organization","volume":"7","author":"Zhang","year":"2017","journal-title":"Sci. Rep."},{"key":"2025101404121494400_JCS264216C56","doi-asserted-by":"publisher","first-page":"224","DOI":"10.1016\/j.devcel.2016.09.029","article-title":"Functional coordination of WAVE and WASP in C. elegans neuroblast migration","volume":"39","author":"Zhu","year":"2016","journal-title":"Dev. Cell"},{"key":"2025101404121494400_JCS264216C57","doi-asserted-by":"publisher","first-page":"2782","DOI":"10.1038\/s41591-024-03197-y","article-title":"Antisense oligonucleotide therapy in an individual with KIF1A-associated neurological disorder","volume":"30","author":"Ziegler","year":"2024","journal-title":"Nat. Med."}],"container-title":["Journal of Cell Science"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.biologists.com\/jcs\/article-pdf\/doi\/10.1242\/jcs.264216\/3665919\/jcs264216.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/jcs\/article-pdf\/doi\/10.1242\/jcs.264216\/3669197\/jcs264216.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/jcs\/article-pdf\/doi\/10.1242\/jcs.264216\/3669197\/jcs264216.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,14]],"date-time":"2025-10-14T08:12:22Z","timestamp":1760429542000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/jcs\/article\/138\/19\/jcs264216\/369517\/Allele-specific-conformational-rescue-of-KIF1A"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,10,1]]},"references-count":58,"journal-issue":{"issue":"19","published-print":{"date-parts":[[2025,10,1]]}},"URL":"https:\/\/doi.org\/10.1242\/jcs.264216","relation":{},"ISSN":["0021-9533","1477-9137"],"issn-type":[{"value":"0021-9533","type":"print"},{"value":"1477-9137","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,10,1]]},"article-number":"jcs264216"}}