{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T02:49:09Z","timestamp":1787021349003,"version":"build-2736575974"},"reference-count":73,"publisher":"The Company of Biologists","issue":"8","license":[{"start":{"date-parts":[[2017,10,15]],"date-time":"2017-10-15T00:00:00Z","timestamp":1508025600000},"content-version":"vor","delay-in-days":183,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/100000069","name":"National Institute of Arthritis and Musculoskeletal and Skin Diseases","doi-asserted-by":"publisher","award":["R01AR059687"],"award-info":[{"award-number":["R01AR059687"]}],"id":[{"id":"10.13039\/100000069","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000062","name":"National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"publisher","award":["P30DK090744"],"award-info":[{"award-number":["P30DK090744"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"publisher","award":["T32GM007499"],"award-info":[{"award-number":["T32GM007499"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["T32HD007149"],"award-info":[{"award-number":["T32HD007149"]}],"id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000001","name":"National Science Foundation","doi-asserted-by":"publisher","award":["DGE-0644492"],"award-info":[{"award-number":["DGE-0644492"]}],"id":[{"id":"10.13039\/100000001","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2017,4,15]]},"abstract":"<jats:p>Cilia are key regulators of animal development and depend on intraflagellar transport (IFT) proteins for their formation and function, yet the roles of individual IFT proteins remain unclear. We examined the Ift56hop mouse mutant and reveal novel insight into the function of IFT56, a poorly understood IFTB protein. Ift56hop mice have normal cilia distribution but display defective cilia structure, including abnormal positioning and number of ciliary microtubule doublets. We show that Ift56hop cilia are unable to accumulate Gli proteins efficiently, resulting in developmental patterning defects in Shh signaling-dependent tissues such as the limb and neural tube. Strikingly, core IFTB proteins are unable to accumulate normally within Ift56hop cilia, including IFT88, IFT81 and IFT27, which are crucial for key processes such as tubulin transport and Shh signaling. IFT56 is required specifically for the IFTB complex, as IFTA components and proteins that rely on IFTA function are unaffected in Ift56hop cilia. These studies define a distinct and novel role for IFT56 in IFTB complex integrity that is crucial for cilia structure and function and, ultimately, animal development.<\/jats:p>","DOI":"10.1242\/dev.143255","type":"journal-article","created":{"date-parts":[[2017,3,6]],"date-time":"2017-03-06T20:10:25Z","timestamp":1488831025000},"page":"1544-1553","source":"Crossref","is-referenced-by-count":37,"title":["IFT56 regulates vertebrate developmental patterning by maintaining IFTB complex integrity and ciliary microtubule architecture"],"prefix":"10.1242","volume":"144","author":[{"given":"Daisy","family":"Xin","sequence":"first","affiliation":[{"name":"Yale University 1 Department of Genetics , , New Haven, CT 06520 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kasey J.","family":"Christopher","sequence":"additional","affiliation":[{"name":"Yale University 1 Department of Genetics , , New Haven, CT 06520 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lewie","family":"Zeng","sequence":"additional","affiliation":[{"name":"Yale University 1 Department of Genetics , , New Haven, CT 06520 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yong","family":"Kong","sequence":"additional","affiliation":[{"name":"Yale University 2 Department of Molecular Biophysics and Biochemistry , , New Haven, CT 06520 , USA"},{"name":"Yale University 3 W.M. Keck Foundation Biotechnology Resource Laboratory , , New Haven, CT 06520 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0915-1329","authenticated-orcid":false,"given":"Scott D.","family":"Weatherbee","sequence":"additional","affiliation":[{"name":"Yale University 1 Department of Genetics , , New Haven, CT 06520 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2024061300015890200_DEV143255C1","doi-asserted-by":"publisher","first-page":"125","DOI":"10.1146\/annurev.genom.7.080505.115610","article-title":"The ciliopathies: an emerging class of human genetic disorders","volume":"7","author":"Badano","year":"2006","journal-title":"Annu. Rev. Genomics Hum. Genet."},{"key":"2024061300015890200_DEV143255C2","doi-asserted-by":"publisher","first-page":"66","DOI":"10.1016\/j.ydbio.2011.09.001","article-title":"Mutations in Traf3ip1 reveal defects in ciliogenesis, embryonic development, and altered cell size regulation","volume":"360","author":"Berbari","year":"2011","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C3","doi-asserted-by":"publisher","first-page":"1009","DOI":"10.1126\/science.1240985","article-title":"Molecular basis of tubulin transport within the cilium by IFT74 and IFT81","volume":"341","author":"Bhogaraju","year":"2013","journal-title":"Science"},{"key":"2024061300015890200_DEV143255C4","doi-asserted-by":"publisher","first-page":"935","DOI":"10.1016\/j.cub.2005.04.059","article-title":"Functional genomics of the cilium, a sensory organelle","volume":"15","author":"Blacque","year":"2005","journal-title":"Curr. Biol."},{"key":"2024061300015890200_DEV143255C5","doi-asserted-by":"publisher","first-page":"2633","DOI":"10.2741\/2871","article-title":"Intraflagellar transport: from molecular characterisation to mechanism","volume":"13","author":"Blacque","year":"2008","journal-title":"Front. Biosci."},{"key":"2024061300015890200_DEV143255C6","doi-asserted-by":"publisher","first-page":"203","DOI":"10.1016\/j.ydbio.2013.05.024","article-title":"Cluap1 localizes preferentially to the base and tip of cilia and is required for ciliogenesis in the mouse embryo","volume":"381","author":"Botilde","year":"2013","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C7","doi-asserted-by":"publisher","first-page":"110","DOI":"10.1016\/j.ydbio.2012.07.017","article-title":"Limb anterior-posterior polarity integrates activator and repressor functions of GLI2 as well as GLI3","volume":"370","author":"Bowers","year":"2012","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C8","doi-asserted-by":"publisher","first-page":"28557","DOI":"10.1074\/jbc.274.40.28557","article-title":"Identification and characterization of Polycystin-2, the PKD2 gene product","volume":"274","author":"Cai","year":"1999","journal-title":"J. Biol. Chem."},{"key":"2024061300015890200_DEV143255C9","doi-asserted-by":"publisher","first-page":"767","DOI":"10.1016\/j.devcel.2007.03.004","article-title":"The graded response to Sonic Hedgehog depends on cilia architecture","volume":"12","author":"Caspary","year":"2007","journal-title":"Dev. Cell"},{"key":"2024061300015890200_DEV143255C10","doi-asserted-by":"publisher","first-page":"407","DOI":"10.1038\/383407a0","article-title":"Cyclopia and defective axial patterning in mice lacking Sonic hedgehog gene function","volume":"383","author":"Chiang","year":"1996","journal-title":"Nature"},{"key":"2024061300015890200_DEV143255C11","doi-asserted-by":"publisher","first-page":"421","DOI":"10.1006\/dbio.2001.0346","article-title":"Manifestation of the limb prepattern: limb development in the absence of sonic hedgehog function","volume":"236","author":"Chiang","year":"2001","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C12","doi-asserted-by":"publisher","first-page":"19096","DOI":"10.1073\/pnas.0910672106","article-title":"Genetic diagnosis by whole exome capture and massively parallel DNA sequencing","volume":"106","author":"Choi","year":"2009","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061300015890200_DEV143255C13","doi-asserted-by":"publisher","first-page":"1427","DOI":"10.1242\/dev.074666","article-title":"Switching on cilia: transcriptional networks regulating ciliogenesis","volume":"141","author":"Choksi","year":"2014","journal-title":"Development"},{"key":"2024061300015890200_DEV143255C14","doi-asserted-by":"publisher","first-page":"382","DOI":"10.1016\/j.ydbio.2012.06.008","article-title":"Forward genetics uncovers Transmembrane protein 107 as a novel factor required for ciliogenesis and Sonic hedgehog signaling","volume":"368","author":"Christopher","year":"2012","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C15","doi-asserted-by":"publisher","first-page":"993","DOI":"10.1083\/jcb.141.4.993","article-title":"Chlamydomonas Kinesin-II\u2013dependent intraflagellar transport (IFT): IFT particles contain proteins required for ciliary assembly in Caenorhabditis elegans sensory neurons","volume":"141","author":"Cole","year":"1998","journal-title":"J. Cell Biol."},{"key":"2024061300015890200_DEV143255C16","doi-asserted-by":"publisher","first-page":"225","DOI":"10.1016\/j.ydbio.2008.10.020","article-title":"Defective ciliogenesis, embryonic lethality and severe impairment of the Sonic Hedgehog pathway caused by inactivation of the mouse complex A intraflagellar transport gene Ift122\/Wdr10, partially overlapping with the DNA repair gene Med1\/Mbd4","volume":"325","author":"Cortellino","year":"2009","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C17","doi-asserted-by":"publisher","first-page":"279","DOI":"10.1016\/j.devcel.2014.09.011","article-title":"IFT27 links the BBSome to IFT for maintenance of the ciliary signaling compartment","volume":"31","author":"Eguether","year":"2014","journal-title":"Dev. Cell"},{"key":"2024061300015890200_DEV143255C18","doi-asserted-by":"publisher","first-page":"451","DOI":"10.1101\/SQB.1997.062.01.053","article-title":"Graded sonic hedgehog signaling and the specification of cell fate in the ventral neural tube","volume":"62","author":"Ericson","year":"1997","journal-title":"Cold Spring Harbor Symp. Quant. Biol."},{"key":"2024061300015890200_DEV143255C19","doi-asserted-by":"publisher","first-page":"880","DOI":"10.1038\/nrm2278","article-title":"When cilia go bad: cilia defects and ciliopathies","volume":"8","author":"Fliegauf","year":"2007","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2024061300015890200_DEV143255C20","doi-asserted-by":"publisher","first-page":"457","DOI":"10.1002\/cm.20346","article-title":"Characterization of mouse IFT complex B","volume":"66","author":"Follit","year":"2009","journal-title":"Cell Motil. Cytoskelet."},{"key":"2024061300015890200_DEV143255C21","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1111\/j.1365-2958.2010.07322.x","article-title":"Biochemical analysis of PIFTC3, the Trypanosoma brucei orthologue of nematode DYF-13, reveals interactions with established and putative intraflagellar transport components","volume":"78","author":"Franklin","year":"2010","journal-title":"Mol. Microbiol."},{"key":"2024061300015890200_DEV143255C22","doi-asserted-by":"publisher","first-page":"331","DOI":"10.1038\/nrg2774","article-title":"The primary cilium: a signalling centre during vertebrate development","volume":"11","author":"Goetz","year":"2010","journal-title":"Nat. Rev. Genet."},{"key":"2024061300015890200_DEV143255C23","doi-asserted-by":"publisher","first-page":"27","DOI":"10.1002\/ajmg.a.31563","article-title":"Two novel point mutations in the long-range SHH enhancer in three families with triphalangeal thumb and preaxial polydactyly","volume":"143A","author":"Gurnett","year":"2007","journal-title":"Am. J. Med. Genet. A"},{"key":"2024061300015890200_DEV143255C24","doi-asserted-by":"publisher","first-page":"e53","DOI":"10.1371\/journal.pgen.0010053","article-title":"Gli2 and Gli3 localize to cilia and require the intraflagellar transport protein polaris for processing and function","volume":"1","author":"Haycraft","year":"2005","journal-title":"PLoS Genet."},{"key":"2024061300015890200_DEV143255C25","doi-asserted-by":"publisher","first-page":"1533","DOI":"10.1056\/NEJMra1010172","article-title":"Ciliopathies","volume":"364","author":"Hildebrandt","year":"2011","journal-title":"N. Engl. J. Med."},{"key":"2024061300015890200_DEV143255C26","doi-asserted-by":"publisher","first-page":"523","DOI":"10.1016\/j.ydbio.2006.09.001","article-title":"Hippi is essential for node cilia assembly and Sonic hedgehog signaling","volume":"300","author":"Houde","year":"2006","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C27","doi-asserted-by":"publisher","first-page":"4","DOI":"10.1186\/2046-2530-1-4","article-title":"Trafficking in and to the primary cilium","volume":"1","author":"Hsiao","year":"2012","journal-title":"Cilia"},{"key":"2024061300015890200_DEV143255C28","doi-asserted-by":"publisher","first-page":"83","DOI":"10.1038\/nature02061","article-title":"Hedgehog signalling in the mouse requires intraflagellar transport proteins","volume":"426","author":"Huangfu","year":"2003","journal-title":"Nature"},{"key":"2024061300015890200_DEV143255C29","doi-asserted-by":"publisher","first-page":"e01566","DOI":"10.7554\/eLife.01566","article-title":"TTC26\/DYF13 is an intraflagellar transport protein required for transport of motility-related proteins into flagella","volume":"3","author":"Ishikawa","year":"2014","journal-title":"Elife"},{"key":"2024061300015890200_DEV143255C30","first-page":"223","article-title":"Hop-sterile, a mutant gene affecting sperm tail development in the mouse","volume":"25","author":"Johnson","year":"1971","journal-title":"J. Embryol. Exp. Morphol."},{"key":"2024061300015890200_DEV143255C31","doi-asserted-by":"publisher","first-page":"10962","DOI":"10.1074\/jbc.M116.713883","article-title":"Overall architecture of the intraflagellar transport (IFT)-B complex containing Cluap1\/IFT38 as an essential component of the IFT-B peripheral subcomplex","volume":"291","author":"Katoh","year":"2016","journal-title":"J. Biol. Chem."},{"key":"2024061300015890200_DEV143255C32","doi-asserted-by":"publisher","first-page":"940","DOI":"10.1016\/j.devcel.2012.04.009","article-title":"IFT25 links the signal-dependent movement of Hedgehog components to intraflagellar transport","volume":"22","author":"Keady","year":"2012","journal-title":"Dev. Cell"},{"key":"2024061300015890200_DEV143255C33","doi-asserted-by":"publisher","first-page":"152","DOI":"10.1016\/j.ygeno.2011.05.009","article-title":"Btrim: a fast, lightweight adapter and quality trimming program for next-generation sequencing technologies","volume":"98","author":"Kong","year":"2011","journal-title":"Genomics"},{"key":"2024061300015890200_DEV143255C34","doi-asserted-by":"publisher","first-page":"5519","DOI":"10.1073\/pnas.90.12.5519","article-title":"A motility in the eukaryotic flagellum unrelated to flagellar beating","volume":"90","author":"Kozminski","year":"1993","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061300015890200_DEV143255C35","doi-asserted-by":"publisher","first-page":"1517","DOI":"10.1083\/jcb.131.6.1517","article-title":"The Chlamydomonas kinesin-like protein FLA10 is involved in motility associated with the flagellar membrane","volume":"131","author":"Kozminski","year":"1995","journal-title":"J. Cell Biol."},{"key":"2024061300015890200_DEV143255C36","doi-asserted-by":"publisher","first-page":"220","DOI":"10.1016\/j.gde.2009.04.008","article-title":"The primary cilium as a cellular signaling center: lessons from disease","volume":"19","author":"Lancaster","year":"2009","journal-title":"Curr. Opin. Genet. Dev."},{"key":"2024061300015890200_DEV143255C37","doi-asserted-by":"publisher","first-page":"248","DOI":"10.1016\/j.ydbio.2015.02.009","article-title":"IFT46 plays an essential role in cilia development","volume":"400","author":"Lee","year":"2015","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C38","doi-asserted-by":"publisher","first-page":"1725","DOI":"10.1093\/hmg\/ddg180","article-title":"A long-range Shh enhancer regulates expression in the developing limb and fin and is associated with preaxial polydactyly","volume":"12","author":"Lettice","year":"2003","journal-title":"Hum. Mol. Genet."},{"key":"2024061300015890200_DEV143255C39","doi-asserted-by":"publisher","first-page":"2078","DOI":"10.1093\/bioinformatics\/btp352","article-title":"The sequence alignment\/map format and SAMtools","volume":"25","author":"Li","year":"2009","journal-title":"Bioinformatics"},{"key":"2024061300015890200_DEV143255C40","doi-asserted-by":"publisher","first-page":"789","DOI":"10.1083\/jcb.201110049","article-title":"The IFT-A complex regulates Shh signaling through cilia structure and membrane protein trafficking","volume":"197","author":"Liem","year":"2012","journal-title":"J. Cell Biol."},{"key":"2024061300015890200_DEV143255C41","doi-asserted-by":"publisher","first-page":"265","DOI":"10.1016\/j.devcel.2014.09.004","article-title":"The intraflagellar transport protein IFT27 promotes BBSome exit from cilia through the GTPase ARL6\/BBS3","volume":"31","author":"Liew","year":"2014","journal-title":"Dev. Cell"},{"key":"2024061300015890200_DEV143255C42","doi-asserted-by":"publisher","first-page":"979","DOI":"10.1038\/79916","article-title":"Specification of ventral neuron types is mediated by an antagonistic interaction between Shh and Gli3","volume":"3","author":"Litingtung","year":"2000","journal-title":"Nat. Neurosci."},{"key":"2024061300015890200_DEV143255C43","doi-asserted-by":"publisher","first-page":"979","DOI":"10.1038\/nature01033","article-title":"Shh and Gli3 are dispensable for limb skeleton formation but regulate digit number and identity","volume":"418","author":"Litingtung","year":"2002","journal-title":"Nature"},{"key":"2024061300015890200_DEV143255C44","doi-asserted-by":"publisher","first-page":"3103","DOI":"10.1242\/dev.01894","article-title":"Mouse intraflagellar transport proteins regulate both the activator and repressor functions of Gli transcription factors","volume":"132","author":"Liu","year":"2005","journal-title":"Development"},{"key":"2024061300015890200_DEV143255C45","doi-asserted-by":"publisher","first-page":"5043","DOI":"10.1073\/pnas.96.9.5043","article-title":"Situs inversus and embryonic ciliary morphogenesis defects in mouse mutants lacking the KIF3A subunit of kinesin-II","volume":"96","author":"Marszalek","year":"1999","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061300015890200_DEV143255C46","doi-asserted-by":"publisher","first-page":"322","DOI":"10.1038\/375322a0","article-title":"Requirement of 19K form of Sonic hedgehog for induction of distinct ventral cell types in CNS explants","volume":"375","author":"Marti","year":"1995","journal-title":"Nature"},{"key":"2024061300015890200_DEV143255C47","doi-asserted-by":"publisher","first-page":"378","DOI":"10.1016\/j.ydbio.2005.08.050","article-title":"Loss of the retrograde motor for IFT disrupts localization of Smo to cilia and prevents the expression of both activator and repressor functions of Gli","volume":"287","author":"May","year":"2005","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C48","doi-asserted-by":"publisher","first-page":"508","DOI":"10.1016\/j.ajhg.2011.03.015","article-title":"Human and mouse mutations in WDR35 cause short-Rib polydactyly syndromes due to abnormal ciliogenesis","volume":"88","author":"Mill","year":"2011","journal-title":"Am. J. Hum. Genet."},{"key":"2024061300015890200_DEV143255C49","volume-title":"Manipulating the Mouse Embryo: A Laboratory Manual","author":"Nagy","year":"2003","edition":"3rd edn."},{"key":"2024061300015890200_DEV143255C50","doi-asserted-by":"publisher","first-page":"663","DOI":"10.1016\/j.cell.2009.10.036","article-title":"Centrioles, centrosomes, and cilia in health and disease","volume":"139","author":"Nigg","year":"2009","journal-title":"Cell"},{"key":"2024061300015890200_DEV143255C51","doi-asserted-by":"publisher","first-page":"443","DOI":"10.1242\/dev.050054","article-title":"Cilia in vertebrate development and disease","volume":"139","author":"Oh","year":"2012","journal-title":"Development"},{"key":"2024061300015890200_DEV143255C52","doi-asserted-by":"publisher","first-page":"2802","DOI":"10.1016\/j.yexcr.2009.06.028","article-title":"Adenylate cyclase regulates elongation of mammalian primary cilia","volume":"315","author":"Ou","year":"2009","journal-title":"Exp. Cell Res."},{"key":"2024061300015890200_DEV143255C53","doi-asserted-by":"publisher","first-page":"2163","DOI":"10.1534\/genetics.108.087114","article-title":"Canine polydactyl mutations with heterogeneous origin in the conserved intronic sequence of LMBR1","volume":"179","author":"Park","year":"2008","journal-title":"Genetics"},{"key":"2024061300015890200_DEV143255C54","doi-asserted-by":"publisher","first-page":"979","DOI":"10.1083\/jcb.141.4.979","article-title":"A dynein light chain is essential for the retrograde particle movement of intraflagellar transport (IFT)","volume":"141","author":"Pazour","year":"1998","journal-title":"J. Cell Biol."},{"key":"2024061300015890200_DEV143255C55","doi-asserted-by":"publisher","first-page":"1993","DOI":"10.1002\/dvdy.21521","article-title":"Assembly of primary cilia","volume":"237","author":"Pedersen","year":"2008","journal-title":"Dev. Dyn."},{"key":"2024061300015890200_DEV143255C56","doi-asserted-by":"publisher","DOI":"10.1101\/gr.080531.108","article-title":"The consensus coding sequence (CCDS) project: Identifying a common protein-coding gene set for the human and mouse genomes (vol 19, pg 1316, 2009)","volume":"19","author":"Pruitt","year":"2009","journal-title":"Genome Res."},{"key":"2024061300015890200_DEV143255C57","doi-asserted-by":"publisher","first-page":"1456","DOI":"10.1073\/pnas.1011410108","article-title":"Intraflagellar transport protein 122 antagonizes Sonic Hedgehog signaling and controls ciliary localization of pathway components","volume":"108","author":"Qin","year":"2011","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061300015890200_DEV143255C58","doi-asserted-by":"publisher","first-page":"372","DOI":"10.1126\/science.1139740","article-title":"Patched1 regulates hedgehog signaling at the primary cilium","volume":"317","author":"Rohatgi","year":"2007","journal-title":"Science"},{"key":"2024061300015890200_DEV143255C59","doi-asserted-by":"publisher","first-page":"813","DOI":"10.1038\/nrm952","article-title":"Intraflagellar transport","volume":"3","author":"Rosenbaum","year":"2002","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2024061300015890200_DEV143255C60","doi-asserted-by":"publisher","first-page":"371","DOI":"10.1016\/S0070-2153(08)00813-2","article-title":"Ciliary dysfunction in developmental abnormalities and diseases","volume":"85","author":"Sharma","year":"2008","journal-title":"Curr. Top. Dev. Biol."},{"key":"2024061300015890200_DEV143255C61","doi-asserted-by":"publisher","first-page":"97","DOI":"10.1016\/S0960-9822(99)80022-0","article-title":"Identification of sonic hedgehog as a candidate gene responsible for the polydactylous mouse mutant Sasquatch","volume":"9","author":"Sharpe","year":"1999","journal-title":"Curr. Biol."},{"key":"2024061300015890200_DEV143255C62","doi-asserted-by":"publisher","first-page":"589","DOI":"10.1136\/jmg.2008.057646","article-title":"Triphalangeal thumb-polysyndactyly syndrome and syndactyly type IV are caused by genomic duplications involving the long range, limb-specific SHH enhancer","volume":"45","author":"Sun","year":"2008","journal-title":"J. Med. Genet."},{"key":"2024061300015890200_DEV143255C63","doi-asserted-by":"publisher","first-page":"e1004689","DOI":"10.1371\/journal.pgen.1004689","article-title":"A mutation in the mouse ttc26 gene leads to impaired hedgehog signaling","volume":"10","author":"Swiderski","year":"2014","journal-title":"PLoS Genet."},{"key":"2024061300015890200_DEV143255C64","doi-asserted-by":"publisher","first-page":"269","DOI":"10.1083\/jcb.201408002","article-title":"Crystal structures of IFT70\/52 and IFT52\/46 provide insight into intraflagellar transport B core complex assembly","volume":"207","author":"Taschner","year":"2014","journal-title":"J. Cell Biol."},{"key":"2024061300015890200_DEV143255C65","doi-asserted-by":"publisher","first-page":"773","DOI":"10.15252\/embj.201593164","article-title":"Intraflagellar transport proteins 172, 80, 57, 54, 38, and 20 form a stable tubulin-binding IFT-B2 complex","volume":"35","author":"Taschner","year":"2016","journal-title":"EMBO J."},{"key":"2024061300015890200_DEV143255C66","doi-asserted-by":"publisher","first-page":"827","DOI":"10.1126\/science.1075620","article-title":"Progression of vertebrate limb development through SHH-mediated counteraction of GLI3","volume":"298","author":"te Welscher","year":"2002","journal-title":"Science"},{"key":"2024061300015890200_DEV143255C67","doi-asserted-by":"publisher","first-page":"403","DOI":"10.1038\/ng.105","article-title":"THM1 negatively modulates mouse sonic hedgehog signal transduction and affects retrograde intraflagellar transport in cilia","volume":"40","author":"Tran","year":"2008","journal-title":"Nat. Genet."},{"key":"2024061300015890200_DEV143255C68","doi-asserted-by":"publisher","first-page":"2651","DOI":"10.1101\/gad.1693008","article-title":"A genome-scale analysis of the cis-regulatory circuitry underlying sonic hedgehog-mediated patterning of the mammalian limb","volume":"22","author":"Vokes","year":"2008","journal-title":"Genes Dev."},{"key":"2024061300015890200_DEV143255C69","doi-asserted-by":"publisher","first-page":"460","DOI":"10.1016\/j.ydbio.2007.02.029","article-title":"The Shh-independent activator function of the full-length Gli3 protein and its role in vertebrate limb digit patterning","volume":"305","author":"Wang","year":"2007","journal-title":"Dev. Biol."},{"key":"2024061300015890200_DEV143255C70","doi-asserted-by":"publisher","first-page":"5557","DOI":"10.1523\/JNEUROSCI.6561-10.2011","article-title":"The type 3 adenylyl cyclase is required for novel object learning and extinction of contextual memory: role of cAMP signaling in primary cilia","volume":"31","author":"Wang","year":"2011","journal-title":"J. Neurosci."},{"key":"2024061300015890200_DEV143255C71","doi-asserted-by":"publisher","first-page":"1039","DOI":"10.1007\/s00467-010-1731-7","article-title":"Ciliopathies: an expanding disease spectrum","volume":"26","author":"Waters","year":"2011","journal-title":"Pediatr. Nephrol."},{"key":"2024061300015890200_DEV143255C72","doi-asserted-by":"publisher","first-page":"4565","DOI":"10.1093\/hmg\/ddp422","article-title":"A mouse model for Meckel syndrome reveals Mks1 is required for ciliogenesis and Hedgehog signaling","volume":"18","author":"Weatherbee","year":"2009","journal-title":"Hum. Mol. Genet."},{"key":"2024061300015890200_DEV143255C73","doi-asserted-by":"publisher","first-page":"3069","DOI":"10.1091\/mbc.E12-01-0019","article-title":"Knockdown of ttc26 disrupts ciliogenesis of the photoreceptor cells and the pronephros in zebrafish","volume":"23","author":"Zhang","year":"2012","journal-title":"Mol. Biol. Cell"}],"container-title":["Development"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1242\/dev.143255","content-type":"unspecified","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dev\/article-pdf\/144\/8\/1544\/3474462\/dev143255.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/journals.biologists.com\/dev\/article-pdf\/doi\/10.1242\/dev.143255\/2053101\/dev_143255v1.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dev\/article-pdf\/144\/8\/1544\/3474462\/dev143255.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,12]],"date-time":"2024-06-12T20:04:06Z","timestamp":1718222646000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dev\/article\/144\/8\/1544\/48404\/IFT56-regulates-vertebrate-developmental"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,4,15]]},"references-count":73,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2017,4,15]]}},"URL":"https:\/\/doi.org\/10.1242\/dev.143255","relation":{},"ISSN":["1477-9129","0950-1991"],"issn-type":[{"value":"1477-9129","type":"electronic"},{"value":"0950-1991","type":"print"}],"subject":[],"published":{"date-parts":[[2017,4,15]]}}}