{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T20:26:30Z","timestamp":1780345590193,"version":"3.54.1"},"reference-count":60,"publisher":"The Company of Biologists","issue":"8","license":[{"start":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T00:00:00Z","timestamp":1693267200000},"content-version":"vor","delay-in-days":28,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100006086","name":"Office of Strategic Coordination","doi-asserted-by":"crossref","award":["U01HG007530"],"award-info":[{"award-number":["U01HG007530"]}],"id":[{"id":"10.13039\/100006086","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100006086","name":"Office of Strategic Coordination","doi-asserted-by":"crossref","award":["U01HG007672"],"award-info":[{"award-number":["U01HG007672"]}],"id":[{"id":"10.13039\/100006086","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000052","name":"NIH Office of the Director","doi-asserted-by":"crossref","award":["U01HG007530"],"award-info":[{"award-number":["U01HG007530"]}],"id":[{"id":"10.13039\/100000052","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000052","name":"NIH Office of the Director","doi-asserted-by":"crossref","award":["U01HG007672"],"award-info":[{"award-number":["U01HG007672"]}],"id":[{"id":"10.13039\/100000052","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R35GM134859"],"award-info":[{"award-number":["R35GM134859"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100007707","name":"University of California, Davis","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007707","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,8,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Hereditary spastic paraplegia (HSP) is a group of degenerative neurological disorders. We identified a variant in human kinesin light chain 4 (KLC4) that is suspected to be associated with autosomal-dominant HSP. How this and other variants relate to pathologies is unknown. We created a humanized Caenorhabditis elegans model in which klc-2 was replaced by human KLC4 (referred to as hKLC4) and assessed the extent to which hKLC4 retained function in the worm. We observed a slight decrease in motility but no nuclear migration defects in the humanized worms, suggesting that hKLC4 retains much of the function of klc-2. Five hKLC4 variants were introduced into the humanized model. The clinical variant led to early lethality, with significant defects in nuclear migration when homozygous and a weak nuclear migration defect when heterozygous, possibly correlating with the clinical finding of late-onset HSP when the proband was heterozygous. Thus, we were able to establish humanized C. elegans as an animal model for HSP and to use it to test the significance of five variants of uncertain significance in the human gene KLC4.<\/jats:p>","DOI":"10.1242\/dmm.050076","type":"journal-article","created":{"date-parts":[[2023,8,11]],"date-time":"2023-08-11T07:53:54Z","timestamp":1691740434000},"source":"Crossref","is-referenced-by-count":5,"title":["A humanized <i>Caenorhabditis elegans<\/i> model of hereditary spastic paraplegia-associated variants in KLC4"],"prefix":"10.1242","volume":"16","author":[{"given":"Selin","family":"G\u00fcm\u00fc\u015fderelio\u011flu","sequence":"first","affiliation":[{"name":"University of California, Davis 1 Department of Molecular and Cellular Biology , , Davis, CA 95616 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren","family":"Resch","sequence":"additional","affiliation":[{"name":"InVivo Biosystems 2 , Eugene, OR 97402 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Trisha","family":"Brock","sequence":"additional","affiliation":[{"name":"InVivo Biosystems 2 , Eugene, OR 97402 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"Undiagnosed Diseases Network","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6180-8906","authenticated-orcid":false,"given":"G. W. Gant","family":"Luxton","sequence":"additional","affiliation":[{"name":"University of California, Davis 1 Department of Molecular and Cellular Biology , , Davis, CA 95616 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heidi","family":"Cope","sequence":"additional","affiliation":[{"name":"Duke University Medical Center 4 Division of Medical Genetics, Department of Pediatrics , , Durham, NC 27710 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Queenie K.-G.","family":"Tan","sequence":"additional","affiliation":[{"name":"Duke University Medical Center 4 Division of Medical Genetics, Department of Pediatrics , , Durham, NC 27710 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher","family":"Hopkins","sequence":"additional","affiliation":[{"name":"InVivo Biosystems 2 , Eugene, OR 97402 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7339-6606","authenticated-orcid":false,"given":"Daniel A.","family":"Starr","sequence":"additional","affiliation":[{"name":"University of California, Davis 1 Department of Molecular and Cellular Biology , , Davis, CA 95616 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2023,8,29]]},"reference":[{"key":"2023082907540423000_DMM050076C1","doi-asserted-by":"publisher","first-page":"248","DOI":"10.1016\/0002-9343(63)90102-5","article-title":"A method and server for predicting damaging missense mutations","volume":"7","author":"Adzhubei","year":"2010","journal-title":"Nat. Methods"},{"key":"2023082907540423000_DMM050076C2","doi-asserted-by":"publisher","first-page":"837","DOI":"10.1534\/genetics.114.169730","article-title":"Efficient marker-free recovery of custom genetic modifications with CRISPR\/Cas9 in Caenorhabditis elegans","volume":"198","author":"Arribere","year":"2014","journal-title":"Genetics"},{"key":"2023082907540423000_DMM050076C3","doi-asserted-by":"publisher","first-page":"496","DOI":"10.1038\/s41419-018-0549-2","article-title":"Kinesin light chain-4 depletion induces apoptosis of radioresistant cancer cells by mitochondrial dysfunction via calcium ion influx article","volume":"9","author":"Baek","year":"2018","journal-title":"Cell Death Dis."},{"key":"2023082907540423000_DMM050076C4","doi-asserted-by":"publisher","first-page":"398","DOI":"10.1038\/s41419-020-2592-z","article-title":"Kinesin light chain 4 as a new target for lung cancer chemoresistance via targeted inhibition of checkpoint kinases in the DNA repair network","volume":"11","author":"Baek","year":"2020","journal-title":"Cell Death Dis."},{"key":"2023082907540423000_DMM050076C5","doi-asserted-by":"publisher","first-page":"206","DOI":"10.1186\/s13023-021-01839-9","article-title":"Model organisms contribute to diagnosis and discovery in the undiagnosed diseases network: current state and a future vision","volume":"16","author":"Baldridge","year":"2021","journal-title":"Orphanet J. Rare Dis."},{"key":"2023082907540423000_DMM050076C6","doi-asserted-by":"publisher","first-page":"763","DOI":"10.1038\/jhg.2015.109","article-title":"Hereditary spastic paraplegia with recessive trait caused by mutation in KLC4 Gene","volume":"60","author":"Bayrakli","year":"2015","journal-title":"J. Hum. Gen."},{"key":"2023082907540423000_DMM050076C7","doi-asserted-by":"publisher","DOI":"10.1895\/wormbook.1.5.2","article-title":"Trans-splicing and operons in C. elegans","author":"Blumenthal","year":"2012","journal-title":"WormBook"},{"key":"2023082907540423000_DMM050076C8","doi-asserted-by":"publisher","first-page":"1951","DOI":"10.1242\/jcs.179788","article-title":"Nuclear migration events throughout development","volume":"129","author":"Bone","year":"2016","journal-title":"J. Cell Sci."},{"key":"2023082907540423000_DMM050076C9","doi-asserted-by":"publisher","first-page":"2853","DOI":"10.1091\/mbc.E14-05-0971","article-title":"The Caenorhabditis elegans SUN protein UNC-84 interacts with lamin to transfer forces from the cytoplasm to the nucleoskeleton during nuclear migration","volume":"25","author":"Bone","year":"2014","journal-title":"Mol. Biol. Cell"},{"key":"2023082907540423000_DMM050076C10","doi-asserted-by":"publisher","first-page":"71","DOI":"10.1080\/01677063.2020.1802723","article-title":"The genetics of Caenorhabditis elegans","volume":"77","author":"Brenner","year":"1974","journal-title":"Genetics"},{"key":"2023082907540423000_DMM050076C11","doi-asserted-by":"publisher","first-page":"e193","DOI":"10.1093\/nar\/gkt805","article-title":"Efficient genome editing in Caenorhabditis elegans by CRISPR-targeted homologous recombination","volume":"41","author":"Chen","year":"2013","journal-title":"Nucleic Acids Res."},{"key":"2023082907540423000_DMM050076C12","doi-asserted-by":"publisher","first-page":"431","DOI":"10.1534\/genetics.118.301775","article-title":"Strategies for efficient genome editing using CRISPR-Cas9","volume":"211","author":"Farboud","year":"2019","journal-title":"Genetics"},{"key":"2023082907540423000_DMM050076C13","doi-asserted-by":"publisher","first-page":"885","DOI":"10.1002\/ajmg.a.61124","article-title":"The burden of rare diseases","volume":"179","author":"Ferreira","year":"2019","journal-title":"Am. J. Med. Genet. A"},{"key":"2023082907540423000_DMM050076C14","doi-asserted-by":"publisher","first-page":"115","DOI":"10.1083\/jcb.201004118","article-title":"Kinesin-1 and dynein at the nuclear envelope mediate the bidirectional migrations of nuclei","volume":"191","author":"Fridolfsson","year":"2010","journal-title":"J. Cell Biol."},{"key":"2023082907540423000_DMM050076C15","doi-asserted-by":"publisher","first-page":"237","DOI":"10.1016\/j.ydbio.2009.12.004","article-title":"UNC-83 Coordinates kinesin-1 and dynein activities at the nuclear envelope during nuclear migration","volume":"338","author":"Fridolfsson","year":"2010","journal-title":"Dev. Biol."},{"key":"2023082907540423000_DMM050076C16","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1007\/978-1-4939-8691-0_13","article-title":"Genetic analysis of nuclear migration and anchorage to study LINC complexes during development of Caenorhabditis elegans","volume":"1840","author":"Fridolfsson","year":"2018","journal-title":"Methods Mol. Biol."},{"key":"2023082907540423000_DMM050076C17","doi-asserted-by":"publisher","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":"Giudice","year":"2014","journal-title":"Arch. Neurol."},{"key":"2023082907540423000_DMM050076C18","doi-asserted-by":"publisher","first-page":"660714","DOI":"10.3389\/fnins.2021.660714","article-title":"Hereditary spastic paraplegia and future therapeutic directions: beneficial effects of small compounds acting on cellular stress","volume":"15","author":"Gumeni","year":"2021","journal-title":"Front. Neurosci."},{"key":"2023082907540423000_DMM050076C19","doi-asserted-by":"publisher","first-page":"707","DOI":"10.1242\/dev.099853","article-title":"Efficient RNA\/Cas9-mediated genome editing in Xenopus Tropicalis","volume":"141","author":"Guo","year":"2014","journal-title":"Development"},{"key":"2023082907540423000_DMM050076C20","doi-asserted-by":"publisher","first-page":"610","DOI":"10.1016\/j.neuron.2010.09.039","article-title":"Molecular motors in neurons: transport mechanisms and roles in brain function, development, and disease","volume":"68","author":"Hirokawa","year":"2010","journal-title":"Neuron"},{"key":"2023082907540423000_DMM050076C21","doi-asserted-by":"publisher","first-page":"101153","DOI":"10.1016\/j.mam.2022.101153","article-title":"Phenotypic screening models for rapid diagnosis of genetic variants and discovery of personalized therapeutics","volume":"91","author":"Hopkins","year":"2022","journal-title":"Mol. Aspects Med."},{"key":"2023082907540423000_DMM050076C22","doi-asserted-by":"publisher","first-page":"877","DOI":"10.1016\/j.ajhg.2016.08.016","article-title":"REVEL: an ensemble method for predicting the pathogenicity of rare missense variants","volume":"99","author":"Ioannidis","year":"2016","journal-title":"Am. J. Hum. Genet."},{"key":"2023082907540423000_DMM050076C23","doi-asserted-by":"publisher","first-page":"147","DOI":"10.1186\/s13023-017-0698-x","article-title":"Nuclear envelopathies: a complex LINC between nuclear envelope and pathology","volume":"12","author":"Janin","year":"2017","journal-title":"Orphanet. J. Rare. Dis."},{"key":"2023082907540423000_DMM050076C24","doi-asserted-by":"publisher","first-page":"583","DOI":"10.1038\/s41586-021-03819-2","article-title":"Highly accurate protein structure prediction with AlphaFold","volume":"596","author":"Jumper","year":"2021","journal-title":"Nature"},{"key":"2023082907540423000_DMM050076C25","doi-asserted-by":"publisher","first-page":"1320","DOI":"10.1095\/biolreprod64.5.1320","article-title":"Kinesin light-chain KLC3 expression in testis is restricted to spermatids","volume":"64","author":"Junco","year":"2001","journal-title":"Biol. Rep."},{"key":"2023082907540423000_DMM050076C26","doi-asserted-by":"publisher","first-page":"434","DOI":"10.1038\/s41586-020-2308-7","article-title":"The mutational constraint spectrum quantified from variation in 141,456 humans","volume":"581","author":"Karczewski","year":"2020","journal-title":"Nature"},{"key":"2023082907540423000_DMM050076C27","doi-asserted-by":"publisher","first-page":"310","DOI":"10.1038\/ng.2892","article-title":"A general framework for estimating the relative pathogenicity of human genetic variants","volume":"46","author":"Kircher","year":"2014","journal-title":"Nat. Genet."},{"key":"2023082907540423000_DMM050076C57","doi-asserted-by":"publisher","first-page":"517","DOI":"10.1016\/S0955-0674(00)00125-3","article-title":"Sorting and transport in C. elegans: a model system with a sequenced genome","volume":"12","author":"Koushika","year":"2000","journal-title":"Curr. Op. Cell Biol."},{"key":"2023082907540423000_DMM050076C28","doi-asserted-by":"publisher","first-page":"dmm049010","DOI":"10.1242\/DMM.049010","article-title":"Caenorhabditis elegans for rare disease modeling and drug discovery: strategies and strengths","volume":"14","author":"Kropp","year":"2021","journal-title":"Dis. Model. Mech."},{"key":"2023082907540423000_DMM050076C29","doi-asserted-by":"publisher","first-page":"1075","DOI":"10.1093\/genetics\/144.3.1075","article-title":"Kinesin mutations cause motor neuron disease phenotypes by disrupting fast axonal transport in drosophila","volume":"144","author":"Kurd","year":"1996","journal-title":"Genetics"},{"key":"2023082907540423000_DMM050076C30","doi-asserted-by":"publisher","first-page":"585","DOI":"10.1016\/s0962-8924(02)02400-5","article-title":"Kinesin motors and disease","volume":"12","author":"Mandelkow","year":"2002","journal-title":"Trends Cell Biol."},{"key":"2023082907540423000_DMM050076C31","doi-asserted-by":"publisher","first-page":"1603","DOI":"10.1016\/j.cub.2011.08.030","article-title":"An EDMD mutation in C. elegans lamin blocks muscle-specific gene relocation and compromises muscle integrity","volume":"21","author":"Mattout","year":"2011","journal-title":"Curr. Biol."},{"key":"2023082907540423000_DMM050076C32","doi-asserted-by":"publisher","first-page":"2725","DOI":"10.1242\/dev.038596","article-title":"UNC-83 Is a nuclear-specific cargo adaptor for kinesin-1-mediated nuclear migration","volume":"136","author":"Meyerzon","year":"2009","journal-title":"Development"},{"key":"2023082907540423000_DMM050076C58","doi-asserted-by":"publisher","first-page":"679","DOI":"10.1038\/s41592-022-01488-1","article-title":"ColabFold: making protein folding accessible to all","volume":"19","author":"Mirdita","year":"2022","journal-title":"Nat. Methods"},{"key":"2023082907540423000_DMM050076C33","doi-asserted-by":"publisher","first-page":"R75","DOI":"10.1186\/gb-2006-7-8-r75","article-title":"Codon usage patterns in nematoda: analysis based on over 25 million codons in thirty-two species","volume":"7","author":"Mitreva","year":"2006","journal-title":"Genome Biol."},{"key":"2023082907540423000_DMM050076C34","doi-asserted-by":"publisher","first-page":"165","DOI":"10.1038\/s41431-019-0508-0","article-title":"Estimating cumulative point prevalence of rare diseases: analysis of the orphanet database","volume":"28","author":"Nguengang Wakap","year":"2020","journal-title":"Eur. J. Hum. Genet."},{"key":"2023082907540423000_DMM050076C35","doi-asserted-by":"publisher","first-page":"52321","DOI":"10.3791\/52321","article-title":"Investigating the spreading and toxicity of prion-like proteins using the metazoan model organism C. elegans","volume":"52321","author":"Nussbaum-Krammer","year":"2015","journal-title":"J. Vis. Exp."},{"key":"2023082907540423000_DMM050076C36","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1534\/genetics.115.179382","article-title":"High efficiency, homology-directed genome editing in Caenorhabditis elegans using CRISPR-Cas9 ribonucleoprotein complexes","volume":"201","author":"Paix","year":"2015","journal-title":"Genetics"},{"key":"2023082907540423000_DMM050076C37","doi-asserted-by":"publisher","first-page":"352","DOI":"10.1016\/j.neurol.2017.03.034","article-title":"Hereditary spastic paraplegia: more than an upper motor neuron disease","volume":"173","author":"Parodi","year":"2017","journal-title":"Rev. Neurol."},{"key":"2023082907540423000_DMM050076C38","doi-asserted-by":"publisher","first-page":"335","DOI":"10.1016\/j.tins.2010.03.006","article-title":"Retrograde axonal transport: pathways to cell death?","volume":"33","author":"Perlson","year":"2010","journal-title":"Trends Neuorosci."},{"key":"2023082907540423000_DMM050076C39","doi-asserted-by":"publisher","first-page":"356","DOI":"10.1126\/science.1234264","article-title":"Structural basis for kinesin-1:cargo recognition","volume":"340","author":"Pernigo","year":"2013","journal-title":"Science"},{"key":"2023082907540423000_DMM050076C59","doi-asserted-by":"publisher","first-page":"70","DOI":"10.1002\/pro.3943","article-title":"UCSF chimerax: structure visualization for researchers, educators, and developers","volume":"30","author":"Pettersen","year":"2021","journal-title":"Protein Science"},{"key":"2023082907540423000_DMM050076C40","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":"2023082907540423000_DMM050076C41","doi-asserted-by":"publisher","first-page":"15395","DOI":"10.1074\/jbc.273.25.15395","article-title":"Two kinesin light chain genes in mice","volume":"273","author":"Rahman","year":"1998","journal-title":"J. Biol. Chem."},{"key":"2023082907540423000_DMM050076C42","doi-asserted-by":"publisher","first-page":"41","DOI":"10.1016\/j.ceb.2007.11.006.C","article-title":"Cargo transport: molecular motors navigate a complex cytoskeleton","volume":"20","author":"Ross","year":"2008","journal-title":"Curr. Opin. Cell Biol."},{"key":"2023082907540423000_DMM050076C43","doi-asserted-by":"publisher","first-page":"2194","DOI":"10.1073\/pnas.0808602106","article-title":"Nesprin 4 is an outer nuclear membrane protein that can induce kinesin-mediated cell polarization","volume":"106","author":"Roux","year":"2009","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023082907540423000_DMM050076C44","doi-asserted-by":"publisher","first-page":"483","DOI":"10.1091\/mbc.E04-07-0553","article-title":"The Caenorhabditis elegans UNC-14 RUN domain protein binds to the kinesin-1 and UNC-16 complex and regulates synaptic vesicle localization","volume":"16","author":"Sakamoto","year":"2005","journal-title":"Mol. Biol. Cell"},{"key":"2023082907540423000_DMM050076C45","doi-asserted-by":"publisher","first-page":"2095","DOI":"10.1242\/jcs.053850","article-title":"The axonal transport of mitochondria","volume":"125","author":"Saxton","year":"2012","journal-title":"J. Cell Sci."},{"key":"2023082907540423000_DMM050076C46","doi-asserted-by":"publisher","first-page":"1136","DOI":"10.1016\/S1474-4422(19)30235-2","article-title":"Hereditary spastic paraplegia: from diagnosis to emerging therapeutic approaches","volume":"18","author":"Shribman","year":"2019","journal-title":"Lancet Neurol."},{"key":"2023082907540423000_DMM050076C47","doi-asserted-by":"publisher","first-page":"452","DOI":"10.1093\/nar\/gks539","article-title":"SIFT Web server: predicting effects of amino acid substitutions on proteins","volume":"40","author":"Sim","year":"2012","journal-title":"Nucleic Acids Res."},{"key":"2023082907540423000_DMM050076C48","doi-asserted-by":"publisher","first-page":"1868","DOI":"10.1056\/nejmoa2035790","article-title":"100,000 Genomes pilot on rare-disease diagnosis in health care \u2014 preliminary report","volume":"385","author":"Smedley","year":"2021","journal-title":"N. Engl. J. Med."},{"key":"2023082907540423000_DMM050076C49","doi-asserted-by":"publisher","first-page":"1323","DOI":"10.1177\/1535370219871965","article-title":"A network of nuclear envelope proteins and cytoskeletal force generators mediates movements of and within nuclei throughout Caenorhabditis elegans Development","volume":"244","author":"Starr","year":"2019","journal-title":"Exp. Biol. Med."},{"key":"2023082907540423000_DMM050076C50","doi-asserted-by":"publisher","first-page":"64","DOI":"10.1016\/0012-1606(83)90201-4","article-title":"The embryonic cell lineage of the nematode Caenorhabditis elegans","volume":"100","author":"Sulston","year":"1983","journal-title":"Dev. Biol."},{"key":"2023082907540423000_DMM050076C51","doi-asserted-by":"publisher","first-page":"974168","DOI":"10.3389\/fcell.2022.974168","article-title":"A nesprin-4\/kinesin-1 cargo model for nuclear positioning in cochlear outer hair cells","volume":"10","author":"Taiber","year":"2022","journal-title":"Front. Cell Dev. Biol."},{"key":"2023082907540423000_DMM050076C52","doi-asserted-by":"publisher","first-page":"39","DOI":"10.1016\/S0092-8674(85)80099-4","article-title":"Identification of a novel force-generating protein, kinesin, involved in microtubule-based motility","volume":"42","author":"Vale","year":"1985","journal-title":"Cell"},{"key":"2023082907540423000_DMM050076C53","doi-asserted-by":"publisher","first-page":"1053","DOI":"10.1083\/jcb.143.4.1053","article-title":"Light chain-dependent regulation of kinesin's interaction with microtubules","volume":"143","author":"Verhey","year":"1998","journal-title":"J. Cell Biol."},{"key":"2023082907540423000_DMM050076C60","doi-asserted-by":"publisher","first-page":"447","DOI":"10.1083\/jcb.200411132","article-title":"Kinesin-1 mediates translocation of the meiotic spindle to the oocyte cortex through KCA-1, a novel cargo adapter","volume":"169","author":"Yang","year":"2005","journal-title":"J. Cell Biol."},{"key":"2023082907540423000_DMM050076C54","doi-asserted-by":"publisher","first-page":"D710","DOI":"10.1093\/nar\/gkv1157","article-title":"Ensembl 2016","volume":"44","author":"Yates","year":"2016","journal-title":"Nucleic Acids Res."},{"key":"2023082907540423000_DMM050076C55","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1016\/j.ydbio.2012.04.026","article-title":"KLC3 Is involved in sperm tail midpiece formation and sperm function","volume":"366","author":"Zhang","year":"2012","journal-title":"Dev. Biol."},{"key":"2023082907540423000_DMM050076C56","doi-asserted-by":"publisher","first-page":"e33943","DOI":"10.1371\/journal.pone.0033943","article-title":"Crystal structures of the tetratricopeptide repeat domains of kinesin light chains: insight into cargo recognition mechanisms","volume":"7","author":"Zhu","year":"2012","journal-title":"PLoS ONE"}],"container-title":["Disease Models &amp; Mechanisms"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.050076\/3102690\/dmm050076.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.050076\/3113398\/dmm050076.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.050076\/3113398\/dmm050076.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T07:54:23Z","timestamp":1693295663000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dmm\/article\/16\/8\/dmm050076\/326587\/A-humanized-Caenorhabditis-elegans-model-of"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,8,1]]},"references-count":60,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2023,8,1]]}},"URL":"https:\/\/doi.org\/10.1242\/dmm.050076","relation":{},"ISSN":["1754-8403","1754-8411"],"issn-type":[{"value":"1754-8403","type":"print"},{"value":"1754-8411","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,8,1]]},"article-number":"dmm050076"}}