{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T06:21:19Z","timestamp":1793254879215,"version":"4.5.0"},"reference-count":123,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2024,7,1]],"date-time":"2024-07-01T00:00:00Z","timestamp":1719792000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2024,7,1]],"date-time":"2024-07-01T00:00:00Z","timestamp":1719792000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T00:00:00Z","timestamp":1716508800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R35 DE027557"],"award-info":[{"award-number":["R35 DE027557"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["F31 DE030664"],"award-info":[{"award-number":["F31 DE030664"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Differentiation"],"published-print":{"date-parts":[[2024,7]]},"DOI":"10.1016\/j.diff.2024.100782","type":"journal-article","created":{"date-parts":[[2024,5,23]],"date-time":"2024-05-23T19:53:22Z","timestamp":1716494002000},"page":"100782","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":2,"special_numbering":"C","title":["The ciliary protein C2cd3 is required for mandibular musculoskeletal tissue patterning"],"prefix":"10.1016","volume":"138","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4895-8296","authenticated-orcid":false,"given":"Evan C.","family":"Brooks","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8039-510X","authenticated-orcid":false,"given":"Simon J.Y.","family":"Han","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5992-5209","authenticated-orcid":false,"given":"Christian Louis","family":"Bonatto Paese","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0003-2766-2688","authenticated-orcid":false,"given":"Amya A.","family":"Lewis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megan","family":"Aarnio-Peterson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6860-6450","authenticated-orcid":false,"given":"Samantha A.","family":"Brugmann","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.diff.2024.100782_bib1","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1093\/oxfordjournals.jhered.a106988","article-title":"Studies with talpid2, an embryonic lethal of the fowl","volume":"51","author":"Abbott","year":"1960","journal-title":"J. Hered."},{"key":"10.1016\/j.diff.2024.100782_bib2","first-page":"1185","article-title":"A 2nd Talpid-like mutation in the fowl","volume":"38","author":"Abbott","year":"1959","journal-title":"Poult. Sci."},{"key":"10.1016\/j.diff.2024.100782_bib3","doi-asserted-by":"crossref","first-page":"97","DOI":"10.1387\/ijdb.170321ja","article-title":"Craniofacial development: discoveries made in the chicken embryo","volume":"62","author":"Abramyan","year":"2018","journal-title":"Int. J. Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib4","doi-asserted-by":"crossref","first-page":"134","DOI":"10.1016\/j.ydbio.2004.05.028","article-title":"Shh and Fgf8 act synergistically to drive cartilage outgrowth during cranial development","volume":"273","author":"Abzhanov","year":"2004","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib5","doi-asserted-by":"crossref","first-page":"1753","DOI":"10.1042\/BST20160241","article-title":"Micrognathia in mouse models of ciliopathies","volume":"44","author":"Adel Al-Lami","year":"2016","journal-title":"Biochem. Soc. Trans."},{"key":"10.1016\/j.diff.2024.100782_bib6","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1038\/s41581-019-0116-9","article-title":"Cellular signalling by primary cilia in development, organ function and disease","volume":"15","author":"Anvarian","year":"2019","journal-title":"Nat. Rev. Nephrol."},{"key":"10.1016\/j.diff.2024.100782_bib7","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0056937","article-title":"Secondary cartilage revealed in a non-avian dinosaur embryo","volume":"8","author":"Bailleul","year":"2013","journal-title":"PLoS One"},{"key":"10.1016\/j.diff.2024.100782_bib8","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0036112","article-title":"First evidence of dinosaurian secondary cartilage in the post-hatching skull of hypacrosaurus stebingeri (dinosauria, ornithischia)","volume":"7","author":"Bailleul","year":"2012","journal-title":"PLoS One"},{"key":"10.1016\/j.diff.2024.100782_bib9","doi-asserted-by":"crossref","first-page":"281","DOI":"10.1002\/ajmg.c.30231","article-title":"Making sense of cilia in disease: the human ciliopathies","volume":"151C","author":"Baker","year":"2009","journal-title":"Am. J. Med. Genet. C Semin. Med. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib10","doi-asserted-by":"crossref","first-page":"391","DOI":"10.1242\/dev.124.2.391","article-title":"Ectopic application of recombinant BMP-2 and BMP-4 can change patterning of developing chick facial primordia","volume":"124","author":"Barlow","year":"1997","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib11","doi-asserted-by":"crossref","first-page":"3779","DOI":"10.1242\/dev.036418","article-title":"A BMP-Shh negative-feedback loop restricts Shh expression during limb development","volume":"136","author":"Bastida","year":"2009","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib12","doi-asserted-by":"crossref","first-page":"85","DOI":"10.1038\/8792","article-title":"Sox9 is required for cartilage formation","volume":"22","author":"Bi","year":"1999","journal-title":"Nat. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib13","doi-asserted-by":"crossref","first-page":"564","DOI":"10.1002\/dvdy.24256","article-title":"Sonic hedgehog from pharyngeal arch 1 epithelium is necessary for early mandibular arch cell survival and later cartilage condensation differentiation","volume":"244","author":"Billmyre","year":"2015","journal-title":"Dev. Dyn."},{"key":"10.1016\/j.diff.2024.100782_bib14","first-page":"89","article-title":"Dental and facial asymmetries: a review","volume":"64","author":"Bishara","year":"1994","journal-title":"Angle Orthod."},{"key":"10.1016\/j.diff.2024.100782_bib15","doi-asserted-by":"crossref","first-page":"126","DOI":"10.1006\/dbio.1995.0010","article-title":"Hedgehog and Bmp genes are coexpressed at many diverse sites of cell-cell interaction in the mouse embryo","volume":"172","author":"Bitgood","year":"1995","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib16","doi-asserted-by":"crossref","first-page":"2680","DOI":"10.1242\/dev.093906","article-title":"Tendon-bone attachment unit is formed modularly by a distinct pool of Scx- and Sox9-positive progenitors","volume":"140","author":"Blitz","year":"2013","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib17","doi-asserted-by":"crossref","first-page":"861","DOI":"10.1016\/j.devcel.2009.10.010","article-title":"Bone Ridge patterning during musculoskeletal assembly is mediated through SCX regulation of Bmp4 at the tendon-skeleton junction","volume":"17","author":"Blitz","year":"2009","journal-title":"Dev. Cell"},{"key":"10.1016\/j.diff.2024.100782_bib18","doi-asserted-by":"crossref","DOI":"10.1242\/dev.186916","article-title":"Development and maintenance of tendons and ligaments","volume":"148","author":"Bobzin","year":"2021","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib19","doi-asserted-by":"crossref","first-page":"1797","DOI":"10.1038\/s41431-018-0222-3","article-title":"Characterization of three ciliopathy pedigrees expands the phenotype associated with biallelic C2CD3 variants","volume":"26","author":"Boczek","year":"2018","journal-title":"Eur. J. Hum. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib20","doi-asserted-by":"crossref","DOI":"10.1242\/dev.194175","article-title":"Ciliopathic micrognathia is caused by aberrant skeletal differentiation and remodeling","volume":"148","author":"Bonatto Paese","year":"2021","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib21","doi-asserted-by":"crossref","DOI":"10.1242\/dmm.049611","article-title":"Pharmacological intervention of the FGF-PTH axis as a potential therapeutic for craniofacial ciliopathies","volume":"15","author":"Bonatto Paese","year":"2022","journal-title":"Dis. Model. Mech."},{"key":"10.1016\/j.diff.2024.100782_bib22","doi-asserted-by":"crossref","first-page":"1358","DOI":"10.1002\/dvdy.318","article-title":"Atavisms in the avian hindlimb and early developmental polarity of the limb","volume":"250","author":"Bonatto Paese","year":"2021","journal-title":"Dev. Dyn."},{"key":"10.1016\/j.diff.2024.100782_bib23","doi-asserted-by":"crossref","first-page":"634","DOI":"10.1016\/j.ajhg.2011.10.001","article-title":"Ciliopathies with skeletal anomalies and renal insufficiency due to mutations in the IFT-A gene WDR19","volume":"89","author":"Bredrup","year":"2011","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib24","doi-asserted-by":"crossref","first-page":"12","DOI":"10.3390\/jdb9020012","article-title":"Mutation in the ciliary protein C2CD3 reveals organ-specific mechanisms of hedgehog signal transduction in avian embryos","volume":"9","author":"Brooks","year":"2021","journal-title":"J. Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib25","doi-asserted-by":"crossref","first-page":"2822","DOI":"10.1093\/hmg\/ddad109","article-title":"Pathogenic RAB34 variants impair primary cilium assembly and cause a novel oral-facial-digital syndrome","volume":"32","author":"Bruel","year":"2023","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib26","doi-asserted-by":"crossref","first-page":"1577","DOI":"10.1093\/hmg\/ddq030","article-title":"A primary cilia-dependent etiology for midline facial disorders","volume":"19","author":"Brugmann","year":"2010","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib27","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1006\/dbio.1999.9321","article-title":"Constitutive activation of sonic hedgehog signaling in the chicken mutant talpid(2): Shh-independent outgrowth and polarizing activity","volume":"212","author":"Caruccio","year":"1999","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib28","doi-asserted-by":"crossref","first-page":"108","DOI":"10.1177\/0022034517732538","article-title":"Ciliopathy protein Tmem107 plays multiple roles in craniofacial development","volume":"97","author":"Cela","year":"2018","journal-title":"J. Dent. Res."},{"key":"10.1016\/j.diff.2024.100782_bib29","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2021.647391","article-title":"Centriolar protein C2cd3 is required for craniofacial development","volume":"9","author":"Chang","year":"2021","journal-title":"Front. Cell Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib30","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1006351","article-title":"Craniofacial ciliopathies reveal specific requirements for GLI proteins during development of the facial midline","volume":"12","author":"Chang","year":"2016","journal-title":"PLoS Genet."},{"key":"10.1016\/j.diff.2024.100782_bib31","doi-asserted-by":"crossref","first-page":"3003","DOI":"10.1242\/dev.105924","article-title":"The cellular and molecular etiology of the craniofacial defects in the avian ciliopathic mutant talpid2","volume":"141","author":"Chang","year":"2014","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib32","doi-asserted-by":"crossref","first-page":"1297","DOI":"10.1177\/0022034518795609","article-title":"Etiology and pathogenesis of hemifacial microsomia","volume":"97","author":"Chen","year":"2018","journal-title":"J. Dent. Res."},{"key":"10.1016\/j.diff.2024.100782_bib33","doi-asserted-by":"crossref","first-page":"3632","DOI":"10.1242\/dev.140137","article-title":"Mapping a multiplexed zoo of mRNA expression","volume":"143","author":"Choi","year":"2016","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib34","doi-asserted-by":"crossref","DOI":"10.1242\/dev.165753","article-title":"Third-generation in situ hybridization chain reaction: multiplexed, quantitative, sensitive, versatile, robust","volume":"145","author":"Choi","year":"2018","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib35","doi-asserted-by":"crossref","first-page":"1806","DOI":"10.1016\/j.celrep.2013.05.020","article-title":"TGF-\u03b2 signaling is associated with endocytosis at the pocket region of the primary cilium","volume":"3","author":"Clement","year":"2013","journal-title":"Cell Rep."},{"key":"10.1016\/j.diff.2024.100782_bib36","doi-asserted-by":"crossref","DOI":"10.1038\/srep24083","article-title":"Mutations in human C2CD3 cause skeletal dysplasia and provide new insights into phenotypic and cellular consequences of altered C2CD3 function","volume":"6","author":"Cort\u00e9s","year":"2016","journal-title":"Sci. Rep."},{"key":"10.1016\/j.diff.2024.100782_bib37","doi-asserted-by":"crossref","first-page":"3445","DOI":"10.1242\/dev.125.17.3445","article-title":"Determination of the identity of the derivatives of the cephalic neural crest: incompatibility between Hox gene expression and lower jaw development","volume":"125","author":"Couly","year":"1998","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib38","doi-asserted-by":"crossref","first-page":"1099","DOI":"10.1242\/dev.121.4.1099","article-title":"Scleraxis: a basic helix-loop-helix protein that prefigures skeletal formation during mouse embryogenesis","volume":"121","author":"Cserjesi","year":"1995","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib39","first-page":"411","article-title":"The segmentation and chondrification of the skull of the duck","volume":"223","author":"de Beer","year":"1934","journal-title":"Philos. Trans. R. Soc. Lond. - Ser. B Contain. Pap. a Biol. Character"},{"key":"10.1016\/j.diff.2024.100782_bib40","doi-asserted-by":"crossref","DOI":"10.3390\/ijerph192114154","article-title":"Mandibular torus as a new index of success for mandibular advancement devices","volume":"19","author":"Diaz de Teran","year":"2022","journal-title":"Int. J. Environ. Res. Public. Health"},{"key":"10.1016\/j.diff.2024.100782_bib41","doi-asserted-by":"crossref","first-page":"167","DOI":"10.1038\/nrg2933","article-title":"Cleft lip and palate: synthesizing genetic and environmental influences","volume":"12","author":"Dixon","year":"2011","journal-title":"Nat. Rev. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib42","doi-asserted-by":"crossref","first-page":"251","DOI":"10.1002\/ar.1092310213","article-title":"Talpid2 mutant chick limb has anteroposterior polarity and altered patterns of programmed cell death","volume":"231","author":"Dvorak","year":"1991","journal-title":"Anat. Rec."},{"key":"10.1016\/j.diff.2024.100782_bib43","doi-asserted-by":"crossref","first-page":"351","DOI":"10.1006\/dbio.2002.0707","article-title":"Fgf4 positively regulates scleraxis and tenascin expression in chick limb tendons","volume":"247","author":"Edom-Vovard","year":"2002","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib44","doi-asserted-by":"crossref","first-page":"28","DOI":"10.1016\/j.ydbio.2018.03.007","article-title":"Sending mixed signals: cilia-dependent signaling during development and disease","volume":"447","author":"Elliott","year":"2019","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib45","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.56450","article-title":"Gli3 utilizes Hand2 to synergistically regulate tissue-specific transcriptional networks","volume":"9","author":"Elliott","year":"2020","journal-title":"Elife"},{"key":"10.1016\/j.diff.2024.100782_bib46","doi-asserted-by":"crossref","first-page":"1323","DOI":"10.1016\/j.mri.2012.05.001","article-title":"3D slicer as an image computing platform for the quantitative imaging network","volume":"30","author":"Fedorov","year":"2012","journal-title":"Magn. Reson. Imaging"},{"key":"10.1016\/j.diff.2024.100782_bib47","doi-asserted-by":"crossref","first-page":"289","DOI":"10.1093\/hmg\/ddn356","article-title":"Convergent extension movements and ciliary function are mediated by ofd1, a zebrafish orthologue of the human oral-facial-digital type 1 syndrome gene","volume":"18","author":"Ferrante","year":"2009","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib48","doi-asserted-by":"crossref","first-page":"12","DOI":"10.1186\/s13630-016-0034-4","article-title":"Update on oral-facial-digital syndromes (OFDS)","volume":"5","author":"Franco","year":"2016","journal-title":"Cilia"},{"key":"10.1016\/j.diff.2024.100782_bib49","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1038\/nrg2774","article-title":"The primary cilium: a signaling center during vertebrate development","volume":"11","author":"Goetz","year":"2010","journal-title":"Nat. Rev. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib50","doi-asserted-by":"crossref","first-page":"301","DOI":"10.1101\/gad.10.3.301","article-title":"Conservation of the hedgehog\/patched signaling pathway from flies to mice: induction of a mouse patched gene by Hedgehog","volume":"10","author":"Goodrich","year":"1996","journal-title":"Genes Dev."},{"key":"10.1016\/j.diff.2024.100782_bib51","doi-asserted-by":"crossref","first-page":"2063","DOI":"10.1242\/dev.125.11.2063","article-title":"Expression and regulation of Lhx6 and Lhx7, a novel subfamily of LIM homeodomain encoding genes, suggests a role in mammalian head development","volume":"125","author":"Grigoriou","year":"1998","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib52","series-title":"Bones and Cartilage: Developmental and Evolutionary Skeletal Biology","author":"Hall","year":"2015"},{"key":"10.1016\/j.diff.2024.100782_bib53","doi-asserted-by":"crossref","first-page":"767","DOI":"10.1002\/ar.1091810408","article-title":"A simple, single-injection method for inducing long-term paralysis in embryonic chicks, and preliminary observations on growth of the tibia","volume":"181","author":"Hall","year":"1975","journal-title":"Anat. Rec."},{"key":"10.1016\/j.diff.2024.100782_bib54","doi-asserted-by":"crossref","first-page":"49","DOI":"10.1002\/jmor.1050880104","article-title":"A series of normal stages in the development of the chick embryo","volume":"88","author":"Hamburger","year":"1951","journal-title":"J. Morphol."},{"key":"10.1016\/j.diff.2024.100782_bib55","doi-asserted-by":"crossref","first-page":"3573","DOI":"10.1016\/j.cell.2021.04.048","article-title":"Integrated analysis of multimodal single-cell data","volume":"184","author":"Hao","year":"2021","journal-title":"Cell"},{"key":"10.1016\/j.diff.2024.100782_bib56","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1016\/j.cub.2005.12.047","article-title":"The development of archosaurian first-generation teeth in a chicken mutant","volume":"16","author":"Harris","year":"2006","journal-title":"Curr. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib57","doi-asserted-by":"crossref","first-page":"3839","DOI":"10.1242\/dev.136242","article-title":"TGF\u03b2 and FGF promote tendon progenitor fate and act downstream of muscle contraction to regulate tendon differentiation during chick limb development","volume":"143","author":"Havis","year":"2016","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib58","doi-asserted-by":"crossref","first-page":"4797","DOI":"10.1242\/dev.01337","article-title":"Regionalisation of early head ectoderm is regulated by endoderm and prepatterns the orofacial epithelium","volume":"131","author":"Haworth","year":"2004","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib59","doi-asserted-by":"crossref","first-page":"244","DOI":"10.1016\/j.ydbio.2006.11.009","article-title":"Sonic hedgehog in the pharyngeal endoderm controls arch pattern via regulation of Fgf8 in head ectoderm","volume":"303","author":"Haworth","year":"2007","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib60","doi-asserted-by":"crossref","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":"10.1016\/j.diff.2024.100782_bib61","doi-asserted-by":"crossref","first-page":"63","DOI":"10.1016\/j.jconrel.2014.05.053","article-title":"Modulating hydrogel crosslink density and degradation to control bone morphogenetic protein delivery and in vivo bone formation","volume":"191","author":"Holloway","year":"2014","journal-title":"J. Control. Release Off. J. Control. Release Soc."},{"key":"10.1016\/j.diff.2024.100782_bib62","doi-asserted-by":"crossref","first-page":"4049","DOI":"10.1242\/dev.029835","article-title":"C2cd3 is required for cilia formation and Hedgehog signaling in mouse","volume":"135","author":"Hoover","year":"2008","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib63","doi-asserted-by":"crossref","first-page":"43","DOI":"10.1016\/j.ydbio.2011.04.019","article-title":"Disrupted dorsal neural tube BMP signaling in the cilia mutant Arl13b hnn stems from abnormal Shh signaling","volume":"355","author":"Horner","year":"2011","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib64","doi-asserted-by":"crossref","first-page":"3727","DOI":"10.1002\/dvdy.21781","article-title":"The effect of BMP signaling on development of the jaw skeleton","volume":"237","author":"Hu","year":"2008","journal-title":"Dev. Dyn."},{"key":"10.1016\/j.diff.2024.100782_bib65","doi-asserted-by":"crossref","first-page":"1749","DOI":"10.1242\/dev.00397","article-title":"A zone of frontonasal ectoderm regulates patterning and growth in the face","volume":"130","author":"Hu","year":"2003","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib66","doi-asserted-by":"crossref","first-page":"11325","DOI":"10.1073\/pnas.0505328102","article-title":"Cilia and Hedgehog responsiveness in the mouse","volume":"102","author":"Huangfu","year":"2005","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.diff.2024.100782_bib67","doi-asserted-by":"crossref","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":"10.1016\/j.diff.2024.100782_bib68","first-page":"1","article-title":"The principles of the Ilizarov method","volume":"48","author":"Ilizarov","year":"1988","journal-title":"Bull. Hosp. Jt. Dis. Orthop. Inst."},{"key":"10.1016\/j.diff.2024.100782_bib69","doi-asserted-by":"crossref","first-page":"1828","DOI":"10.1016\/j.ajhg.2022.08.009","article-title":"Mutations in SCNM1 cause orofaciodigital syndrome due to minor intron splicing defects affecting primary cilia","volume":"109","author":"Iturrate","year":"2022","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib70","doi-asserted-by":"crossref","first-page":"937","DOI":"10.1101\/gad.1190304","article-title":"Hedgehog signaling in the neural crest cells regulates the patterning and growth of facial primordia","volume":"18","author":"Jeong","year":"2004","journal-title":"Genes Dev."},{"key":"10.1016\/j.diff.2024.100782_bib71","doi-asserted-by":"crossref","first-page":"1088","DOI":"10.1038\/s41467-021-21246-9","article-title":"Inference and analysis of cell-cell communication using CellChat","volume":"12","author":"Jin","year":"2021","journal-title":"Nat. Commun."},{"key":"10.1016\/j.diff.2024.100782_bib72","doi-asserted-by":"crossref","first-page":"513","DOI":"10.1038\/nrd4233","article-title":"Can we safely target the WNT pathway?","volume":"13","author":"Kahn","year":"2014","journal-title":"Nat. Rev. Drug Discov."},{"key":"10.1016\/j.diff.2024.100782_bib73","doi-asserted-by":"crossref","first-page":"84","DOI":"10.3390\/genes14010084","article-title":"Dental anomalies in ciliopathies: lessons from patients with BBS2, BBS7, and EVC2 mutations","volume":"14","author":"Kantaputra","year":"2023","journal-title":"Genes"},{"key":"10.1016\/j.diff.2024.100782_bib74","doi-asserted-by":"crossref","first-page":"21666","DOI":"10.1073\/pnas.0912180106","article-title":"Gli2 trafficking links Hedgehog-dependent activation of Smoothened in the primary cilium to transcriptional activation in the nucleus","volume":"106","author":"Kim","year":"2009","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.diff.2024.100782_bib75","doi-asserted-by":"crossref","first-page":"317","DOI":"10.1111\/joa.13096","article-title":"Ift88 is involved in mandibular development","volume":"236","author":"Kitamura","year":"2020","journal-title":"J. Anat."},{"key":"10.1016\/j.diff.2024.100782_bib76","series-title":"Evolution of the House Mouse, Cambridge Studies in Morphology and Molecules: New Paradigms in Evolutionary Bio","first-page":"135","article-title":"Development of the mouse mandible: a model system for complex morphological structures","author":"Klingenberg","year":"2012"},{"key":"10.1016\/j.diff.2024.100782_bib77","doi-asserted-by":"crossref","first-page":"273","DOI":"10.1016\/j.ydbio.2007.07.018","article-title":"Kinesin-2 controls development and patterning of the vertebrate skeleton by hedgehog- and gli3-dependent mechanisms","volume":"309","author":"Kolpakova-Hart","year":"2007","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib78","doi-asserted-by":"crossref","first-page":"3229","DOI":"10.1242\/dev.122.10.3229","article-title":"Rhombencephalic neural crest segmentation is preserved throughout craniofacial ontogeny","volume":"122","author":"K\u00f6ntges","year":"1996","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib79","doi-asserted-by":"crossref","first-page":"1151","DOI":"10.1083\/jcb.102.4.1151","article-title":"Collagen gene expression during limb cartilage differentiation","volume":"102","author":"Kosher","year":"1986","journal-title":"J. Cell Biol."},{"key":"10.1016\/j.diff.2024.100782_bib80","doi-asserted-by":"crossref","first-page":"229","DOI":"10.1016\/j.ydbio.2013.04.026","article-title":"The widely used Wnt1-Cre transgene causes developmental phenotypes by ectopic activation of Wnt signaling","volume":"379","author":"Lewis","year":"2013","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib81","doi-asserted-by":"crossref","first-page":"6361","DOI":"10.1242\/dev.00850","article-title":"Ablation of specific expression domains reveals discrete functions of ectoderm- and endoderm-derived FGF8 during cardiovascular and pharyngeal development","volume":"130","author":"Macatee","year":"2003","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib82","doi-asserted-by":"crossref","first-page":"S77","DOI":"10.4103\/jpn.JPN_17_22","article-title":"Evaluation and management of nonsyndromic craniosynostosis","volume":"17","author":"Marbate","year":"2022","journal-title":"J. Pediatr. Neurosci."},{"key":"10.1016\/j.diff.2024.100782_bib83","doi-asserted-by":"crossref","first-page":"1225","DOI":"10.1242\/dev.122.4.1225","article-title":"Conservation in hedgehog signaling: induction of a chicken patched homolog by Sonic hedgehog in the developing limb","volume":"122","author":"Marigo","year":"1996","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib84","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1097\/00006534-199289010-00001","article-title":"Lengthening the human mandible by gradual distraction","volume":"89","author":"McCarthy","year":"1992","journal-title":"Plast. Reconstr. Surg."},{"key":"10.1016\/j.diff.2024.100782_bib85","doi-asserted-by":"crossref","DOI":"10.1101\/cshperspect.a022095","article-title":"Regulation of TGF-\u03b2 family signaling by inhibitory smads","volume":"9","author":"Miyazawa","year":"2017","journal-title":"Cold Spring Harb. Perspect. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib86","doi-asserted-by":"crossref","first-page":"e38","DOI":"10.1002\/cpcy.38","article-title":"Clearing for deep tissue imaging","volume":"86","author":"Muntifering","year":"2018","journal-title":"Curr. Protoc. Cytom."},{"key":"10.1016\/j.diff.2024.100782_bib87","doi-asserted-by":"crossref","first-page":"368","DOI":"10.1071\/ZO9630368","article-title":"Adventitious (Secondary) cartilage in the chick, and the development of certain bones and articulation in the chick skull","volume":"11","author":"Murray","year":"1963","journal-title":"Aust. J. Zool."},{"key":"10.1016\/j.diff.2024.100782_bib88","doi-asserted-by":"crossref","DOI":"10.1101\/pdb.prot5169","article-title":"Alcian blue staining of the mouse fetal cartilaginous skeleton","volume":"2009","author":"Nagy","year":"2009","journal-title":"Cold Spring Harb. Protoc."},{"key":"10.1016\/j.diff.2024.100782_bib89","doi-asserted-by":"crossref","first-page":"247","DOI":"10.1016\/S0092-8674(00)80333-5","article-title":"Antagonistic interactions between FGF and BMP signaling pathways: a mechanism for positioning the sites of tooth formation","volume":"90","author":"Neub\u00fcser","year":"1997","journal-title":"Cell"},{"key":"10.1016\/j.diff.2024.100782_bib90","first-page":"795","article-title":"Convergence of the BMP and EGF signaling pathways on Smad1 in the regulation of chondrogenesis","volume":"43","author":"Nonaka","year":"1999","journal-title":"Int. J. Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib91","doi-asserted-by":"crossref","first-page":"47","DOI":"10.1016\/S0960-9822(01)00631-5","article-title":"Opponent activities of Shh and BMP signaling during floor plate induction in vivo","volume":"12","author":"Patten","year":"2002","journal-title":"Curr. Biol. CB"},{"key":"10.1016\/j.diff.2024.100782_bib92","doi-asserted-by":"crossref","DOI":"10.3389\/fgene.2022.871927","article-title":"Meckel's cartilage in mandibular development and dysmorphogenesis","volume":"13","author":"Pitirri","year":"2022","journal-title":"Front. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib93","doi-asserted-by":"crossref","first-page":"261","DOI":"10.1016\/j.ydbio.2010.11.012","article-title":"Ecotdermal Wnt\/\u03b2-Catenin signaling shapes the mouse face","volume":"349","author":"Reid","year":"2011","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib94","doi-asserted-by":"crossref","first-page":"533","DOI":"10.1038\/nrm.2017.60","article-title":"Genes and molecular pathways underpinning ciliopathies","volume":"18","author":"Reiter","year":"2017","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"10.1016\/j.diff.2024.100782_bib95","doi-asserted-by":"crossref","DOI":"10.1242\/dev.170241","article-title":"FGF signaling patterns cell fate at the interface between tendon and bone","volume":"146","author":"Roberts","year":"2019","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib96","doi-asserted-by":"crossref","first-page":"329","DOI":"10.1242\/dev.117.1.329","article-title":"Cellular patterning of fast and slow fibres in the intermandibularis muscle of chick embryos","volume":"117","author":"Robson","year":"1993","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib97","doi-asserted-by":"crossref","first-page":"944","DOI":"10.1002\/ca.22275","article-title":"Origin of the torus mandibularis: an embryological hypothesis","volume":"26","author":"Rodr\u00edguez-V\u00e1zquez","year":"2013","journal-title":"Clin. Anat. N. Y. N"},{"key":"10.1016\/j.diff.2024.100782_bib98","doi-asserted-by":"crossref","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":"10.1016\/j.diff.2024.100782_bib99","doi-asserted-by":"crossref","first-page":"103","DOI":"10.1007\/s004410051271","article-title":"From head to toe: conservation of molecular signals regulating limb and craniofacial morphogenesis","volume":"296","author":"Schneider","year":"1999","journal-title":"Cell Tissue Res."},{"key":"10.1016\/j.diff.2024.100782_bib100","doi-asserted-by":"crossref","DOI":"10.1101\/cshperspect.a028258","article-title":"Discovery, diagnosis, and etiology of craniofacial ciliopathies","volume":"9","author":"Schock","year":"2017","journal-title":"Cold Spring Harb. Perspect. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib101","first-page":"855","article-title":"Using the avian mutant talpid2 as a disease model for understanding the oral-facial phenotypes of oral-facial-digital syndrome","volume":"8","author":"Schock","year":"2015","journal-title":"Dis. Model. Mech."},{"key":"10.1016\/j.diff.2024.100782_bib102","doi-asserted-by":"crossref","first-page":"3855","DOI":"10.1242\/dev.128.19.3855","article-title":"Analysis of the tendon cell fate using Scleraxis, a specific marker for tendons and ligaments","volume":"128","author":"Schweitzer","year":"2001","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib103","doi-asserted-by":"crossref","first-page":"2807","DOI":"10.1242\/dev.047498","article-title":"Connecting muscles to tendons: tendons and musculoskeletal development in flies and vertebrates","volume":"137","author":"Schweitzer","year":"2010","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib104","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1002\/(SICI)1097-0177(200004)217:4<401::AID-DVDY7>3.0.CO;2-D","article-title":"Positionally-dependent chondrogenesis induced by BMP4 is co-regulated by sox9 and msx2","volume":"217","author":"Semba","year":"2000","journal-title":"Dev. Dyn."},{"key":"10.1016\/j.diff.2024.100782_bib105","doi-asserted-by":"crossref","first-page":"273","DOI":"10.1007\/s00429-004-0393-2","article-title":"Runx2-deficient mice lack mandibular condylar cartilage and have deformed Meckel's cartilage","volume":"208","author":"Shibata","year":"2004","journal-title":"Anat. Embryol."},{"key":"10.1016\/j.diff.2024.100782_bib106","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1006\/dbio.2000.9932","article-title":"Ectodermally derived FGF8 defines the maxillomandibular region in the early chick embryo: epithelial\u2013mesenchymal interactions in the specification of the craniofacial ectomesenchyme","volume":"228","author":"Shigetani","year":"2000","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib107","doi-asserted-by":"crossref","first-page":"28","DOI":"10.1016\/j.ydbio.2011.05.003","article-title":"Mesenchymal and mechanical mechanisms of secondary cartilage induction","volume":"356","author":"Solem","year":"2011","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib108","doi-asserted-by":"crossref","first-page":"2280","DOI":"10.1242\/dev.096354","article-title":"Scx+\/Sox9+ progenitors contribute to the establishment of the junction between cartilage and tendon\/ligament","volume":"140","author":"Sugimoto","year":"2013","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib109","doi-asserted-by":"crossref","first-page":"3060","DOI":"10.1038\/s41467-023-38647-7","article-title":"Directionality of developing skeletal muscles is set by mechanical forces","volume":"14","author":"Sunadome","year":"2023","journal-title":"Nat. Commun."},{"key":"10.1016\/j.diff.2024.100782_bib110","doi-asserted-by":"crossref","first-page":"821","DOI":"10.3389\/fcell.2020.00821","article-title":"Diverse fate of an enigmatic structure: 200 Years of Meckel's cartilage","volume":"8","author":"Svandova","year":"2020","journal-title":"Front. Cell Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib111","doi-asserted-by":"crossref","first-page":"623","DOI":"10.1016\/j.devcel.2013.05.021","article-title":"Fuz mutant mice reveal shared mechanisms between ciliopathies and FGF-related syndromes","volume":"25","author":"Tabler","year":"2013","journal-title":"Dev. Cell"},{"key":"10.1016\/j.diff.2024.100782_bib112","doi-asserted-by":"crossref","first-page":"905","DOI":"10.1038\/ng.3031","article-title":"The oral-facial-digital syndrome gene C2CD3 encodes a positive regulator of centriole elongation","volume":"46","author":"Thauvin-Robinet","year":"2014","journal-title":"Nat. Genet."},{"key":"10.1016\/j.diff.2024.100782_bib113","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1126\/science.282.5391.1136","article-title":"Transformation of tooth type induced by inhibition of BMP signaling","volume":"282","author":"Tucker","year":"1998","journal-title":"Science"},{"key":"10.1016\/j.diff.2024.100782_bib114","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1242\/dev.126.1.51","article-title":"Fgf-8 determines rostral-caudal polarity in the first branchial arch","volume":"126","author":"Tucker","year":"1999","journal-title":"Development"},{"key":"10.1016\/j.diff.2024.100782_bib115","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1016\/S0092-8674(05)80083-2","article-title":"Identification of BMP-4 as a signal mediating secondary induction between epithelial and mesenchymal tissues during early tooth development","volume":"75","author":"Vainio","year":"1993","journal-title":"Cell"},{"key":"10.1016\/j.diff.2024.100782_bib116","doi-asserted-by":"crossref","first-page":"22","DOI":"10.1016\/j.jmoldx.2011.08.002","article-title":"RNAscope: a novel in situ RNA analysis platform for formalin-fixed, paraffin-embedded tissues","volume":"14","author":"Wang","year":"2012","journal-title":"J. Mol. Diagn."},{"key":"10.1016\/j.diff.2024.100782_bib117","doi-asserted-by":"crossref","first-page":"1910","DOI":"10.1128\/MCB.01089-09","article-title":"Kinetics of hedgehog-dependent full-length Gli3 accumulation in primary cilia and subsequent degradation","volume":"30","author":"Wen","year":"2010","journal-title":"Mol. Cell Biol."},{"key":"10.1016\/j.diff.2024.100782_bib118","doi-asserted-by":"crossref","first-page":"2759","DOI":"10.1073\/pnas.1018823108","article-title":"Primary cilia membrane assembly is initiated by Rab11 and transport protein particle II (TRAPPII) complex-dependent trafficking of Rabin8 to the centrosome","volume":"108","author":"Westlake","year":"2011","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.diff.2024.100782_bib119","doi-asserted-by":"crossref","first-page":"S219","DOI":"10.1016\/j.ydbio.2018.05.002","article-title":"FGF and TGF\u03b2 signaling link form and function during jaw development and evolution","volume":"444","author":"Woronowicz","year":"2018","journal-title":"Dev. Biol."},{"key":"10.1016\/j.diff.2024.100782_bib120","doi-asserted-by":"crossref","first-page":"607","DOI":"10.1016\/0092-8674(89)90583-7","article-title":"Myogenin, a factor regulating myogenesis, has a domain homologous to MyoD","volume":"56","author":"Wright","year":"1989","journal-title":"Cell"},{"key":"10.1016\/j.diff.2024.100782_bib121","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.40315","article-title":"Hedgehog signaling patterns the oral-aboral axis of the mandibular arch","volume":"8","author":"Xu","year":"2019","journal-title":"Elife"},{"key":"10.1016\/j.diff.2024.100782_bib122","doi-asserted-by":"crossref","first-page":"2164","DOI":"10.1073\/pnas.1318737111","article-title":"C2cd3 is critical for centriolar distal appendage assembly and ciliary vesicle docking in mammals","volume":"111","author":"Ye","year":"2014","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.diff.2024.100782_bib123","article-title":"Fuz regulates craniofacial development through tissue specific responses to signaling factors","volume":"6","author":"Zhang","year":"2011","journal-title":"PLoS One"}],"container-title":["Differentiation"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0301468124000380?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0301468124000380?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2024,7,4]],"date-time":"2024-07-04T10:48:18Z","timestamp":1720090098000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0301468124000380"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7]]},"references-count":123,"alternative-id":["S0301468124000380"],"URL":"https:\/\/doi.org\/10.1016\/j.diff.2024.100782","relation":{"has-preprint":[{"id-type":"doi","id":"10.1101\/2024.02.23.581730","asserted-by":"object"}]},"ISSN":["0301-4681"],"issn-type":[{"value":"0301-4681","type":"print"}],"subject":[],"published":{"date-parts":[[2024,7]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"The ciliary protein C2cd3 is required for mandibular musculoskeletal tissue patterning","name":"articletitle","label":"Article Title"},{"value":"Differentiation","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.diff.2024.100782","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2024 The Authors. Published by Elsevier B.V. on behalf of International Society of Differentiation.","name":"copyright","label":"Copyright"}],"article-number":"100782"}}