{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T11:23:06Z","timestamp":1792754586976,"version":"4.5.0"},"reference-count":233,"publisher":"MDPI AG","issue":"3","license":[{"start":{"date-parts":[[2020,9,9]],"date-time":"2020-09-09T00:00:00Z","timestamp":1599609600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["R01DE025827"],"award-info":[{"award-number":["R01DE025827"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["T32DE007057"],"award-info":[{"award-number":["T32DE007057"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JDB"],"abstract":"<jats:p>Craniofacial anomalies are among the most common of birth defects. The pathogenesis of craniofacial anomalies frequently involves defects in the migration, proliferation, and fate of neural crest cells destined for the craniofacial skeleton. Genetic mutations causing deficient cranial neural crest migration and proliferation can result in Treacher Collins syndrome, Pierre Robin sequence, and cleft palate. Defects in post-migratory neural crest cells can result in pre- or post-ossification defects in the developing craniofacial skeleton and craniosynostosis (premature fusion of cranial bones\/cranial sutures). The coronal suture is the most frequently fused suture in craniosynostosis syndromes. It exists as a biological boundary between the neural crest-derived frontal bone and paraxial mesoderm-derived parietal bone. The objective of this review is to frame our current understanding of neural crest cells in craniofacial development, craniofacial anomalies, and the pathogenesis of coronal craniosynostosis. We will also discuss novel approaches for advancing our knowledge and developing prevention and\/or treatment strategies for craniofacial tissue regeneration and craniosynostosis.<\/jats:p>","DOI":"10.3390\/jdb8030018","type":"journal-article","created":{"date-parts":[[2020,9,9]],"date-time":"2020-09-09T09:01:09Z","timestamp":1599642069000},"page":"18","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":63,"title":["Cranial Neural Crest Cells and Their Role in the Pathogenesis of Craniofacial Anomalies and Coronal Craniosynostosis"],"prefix":"10.3390","volume":"8","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5981-3482","authenticated-orcid":false,"given":"Erica M.","family":"Siismets","sequence":"first","affiliation":[{"name":"Oral Health Sciences PhD Program, School of Dentistry, University of Michigan, Ann Arbor, MI 48109-1078, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2632-0841","authenticated-orcid":false,"given":"Nan E.","family":"Hatch","sequence":"additional","affiliation":[{"name":"Department of Orthodontics and Pediatric Dentistry, School of Dentistry, University of Michigan, Ann Arbor, MI 48109-1078, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2020,9,9]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1950","DOI":"10.1002\/jor.23209","article-title":"RNA-seq analysis of clinical-grade osteochondral allografts reveals activation of early response genes","volume":"34","author":"Lin","year":"2016","journal-title":"J. Orthop. Res."},{"key":"ref_2","first-page":"21","article-title":"Environment and genetics in the etiology of cleft lip and cleft palate with reference to the role of folic acid","volume":"24","author":"Bianchi","year":"2000","journal-title":"Epidemiol. Prev."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"101528","DOI":"10.1016\/j.scr.2019.101528","article-title":"Pharmacological exposures may precipitate craniosynostosis through targeted stem cell depletion","volume":"40","author":"Durham","year":"2019","journal-title":"Stem Cell Res."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"959","DOI":"10.1177\/0022034519851786","article-title":"Regulatory Mechanisms of Soft Palate Development and Malformations","volume":"98","author":"Li","year":"2019","journal-title":"J. Dent. Res."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"13403","DOI":"10.1073\/pnas.0603730103","article-title":"Tcof1\/Treacle is required for neural crest cell formation and proliferation deficiencies that cause craniofacial abnormalities","volume":"103","author":"Dixon","year":"2006","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_6","first-page":"3734","article-title":"Disruption of the ERK\/MAPK pathway in neural crest cells as a potential cause of Pierre Robin sequence","volume":"142","author":"Parada","year":"2015","journal-title":"Development"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1159\/000491004","article-title":"Mouse Models of Syndromic Craniosynostosis","volume":"10","author":"Lee","year":"2018","journal-title":"Mol. Syndr."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1479","DOI":"10.7150\/ijbs.22373","article-title":"Fibroblast Growth Factor Receptor 2 (FGFR2) Mutation Related Syndromic Craniosynostosis","volume":"13","author":"Azoury","year":"2017","journal-title":"Int. J. Biol. Sci."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"359","DOI":"10.1016\/j.ajhg.2015.07.006","article-title":"A Genetic-Pathophysiological Framework for Craniosynostosis","volume":"97","author":"Twigg","year":"2015","journal-title":"Am. J. Hum. Genet."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1346","DOI":"10.1242\/dev.076729","article-title":"Regulation of cranial morphogenesis and cell fate at the neural crest-mesoderm boundary by engrailed 1","volume":"139","author":"Deckelbaum","year":"2012","journal-title":"Development"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"1319","DOI":"10.1093\/hmg\/ddl052","article-title":"Cell mixing at a neural crest-mesoderm boundary and deficient ephrin-Eph signaling in the pathogenesis of craniosynostosis","volume":"15","author":"Merrill","year":"2006","journal-title":"Hum. Mol. Genet."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"242","DOI":"10.1242\/dev.105445","article-title":"Establishing neural crest identity: A gene regulatory recipe","volume":"142","author":"Bronner","year":"2015","journal-title":"Development"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"43","DOI":"10.1016\/j.ceb.2020.05.001","article-title":"Epithelial-to-mesenchymal transition and different migration strategies as viewed from the neural crest","volume":"66","author":"Piacentino","year":"2020","journal-title":"Curr. Opin. Cell Biol."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"835","DOI":"10.1083\/jcb.201305050","article-title":"Sip1 mediates an E-cadherin-to-N-cadherin switch during cranial neural crest EMT","volume":"203","author":"Rogers","year":"2013","journal-title":"J. Cell Biol."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"1126","DOI":"10.1038\/s41467-018-03512-5","article-title":"Glycogen synthase kinase 3 controls migration of the neural crest lineage in mouse and Xenopus","volume":"9","author":"Malagon","year":"2018","journal-title":"Nat. Commun."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"1542","DOI":"10.1038\/ncomms2543","article-title":"Interactions between Twist and other core epithelial\u2013mesenchymal transition factors are controlled by GSK3-mediated phosphorylation","volume":"4","author":"Lander","year":"2013","journal-title":"Nat. Commun."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"76","DOI":"10.1038\/35000025","article-title":"The transcription factor snail controls epithelial\u2013mesenchymal transitions by repressing E-cadherin expression","volume":"2","author":"Cano","year":"2000","journal-title":"Nat. Cell Biol."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"715","DOI":"10.1016\/j.tig.2017.07.015","article-title":"Regulatory Logic Underlying Diversification of the Neural Crest","volume":"33","author":"Martik","year":"2017","journal-title":"Trends Genet."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"431","DOI":"10.4161\/cam.4.3.12138","article-title":"Regulation of cadherin expression in the chicken neural crest by the Wnt\/beta-catenin signaling pathway","volume":"4","author":"Chalpe","year":"2010","journal-title":"Cell Adhes. Migr."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"162","DOI":"10.1016\/j.ydbio.2012.01.028","article-title":"Matrix metalloproteinase 9\/gelatinase B is required for neural crest cell migration","volume":"364","author":"Kosonovsky","year":"2012","journal-title":"Dev. Biol."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"5240","DOI":"10.1096\/fj.201901217RR","article-title":"Conserved role of matrix metalloproteases 2 and 9 in promoting the migration of neural crest cells in avian and mammalian embryos","volume":"34","author":"Hanael","year":"2020","journal-title":"FASEB J."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"4072","DOI":"10.1091\/mbc.e14-05-0970","article-title":"GSK3 and Polo-like kinase regulate ADAM13 function during cranial neural crest cell migration","volume":"25","author":"Abbruzzese","year":"2014","journal-title":"Mol. Biol. Cell"},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1038\/nature05557","article-title":"Chemical rescue of cleft palate and midline defects in conditional GSK-3beta mice","volume":"446","author":"Liu","year":"2007","journal-title":"Nature"},{"key":"ref_24","doi-asserted-by":"crossref","unstructured":"Szabo-Rogers, H., Yakob, W., and Liu, K.J. (2016). Frontal Bone Insufficiency in Gsk3beta Mutant Mice. PLoS ONE, 11.","DOI":"10.1371\/journal.pone.0149604"},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"683","DOI":"10.1016\/j.semcdb.2005.06.007","article-title":"Specification of neural crest cell formation and migration in mouse embryos","volume":"16","author":"Trainor","year":"2005","journal-title":"Semin. Cell Dev. Biol."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"184","DOI":"10.1016\/j.ydbio.2020.02.010","article-title":"Visualizing mesoderm and neural crest cell dynamics during chick head morphogenesis","volume":"461","author":"McKinney","year":"2020","journal-title":"Dev. Biol."},{"key":"ref_27","unstructured":"Trainor, P.A. (2014). Chapter 7\u2014Neural Crest Cells in Craniofacial Skeletal Development. Neural Crest Cells, Academic Press."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1186\/2041-9139-3-24","article-title":"Developmental and evolutionary origins of the pharyngeal apparatus","volume":"3","author":"Graham","year":"2012","journal-title":"EvoDevo"},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"2935","DOI":"10.1242\/dev.081471","article-title":"Multiscale mechanisms of cell migration during development: Theory and experiment","volume":"139","author":"McLennan","year":"2012","journal-title":"Development"},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"114","DOI":"10.1016\/j.ydbio.2009.12.022","article-title":"Vascular endothelial growth factor (VEGF) regulates cranial neural crest migration in vivo","volume":"339","author":"McLennan","year":"2010","journal-title":"Dev. Biol."},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"170","DOI":"10.1002\/(SICI)1097-0177(200002)217:2<170::AID-DVDY4>3.0.CO;2-9","article-title":"Chemotactic migration of mesencephalic neural crest cells in the mouse","volume":"217","author":"Kubota","year":"2000","journal-title":"Dev. Dyn."},{"key":"ref_32","doi-asserted-by":"crossref","unstructured":"Dunkel, H., Chaverra, M., Bradley, R., and Lefcort, F. (2020). FGF signaling is required for chemokinesis and ventral migration of trunk neural crest cells. Dev. Dyn.","DOI":"10.1002\/dvdy.190"},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"3136","DOI":"10.1101\/gad.13.23.3136","article-title":"Cre-mediated gene inactivation demonstrates that FGF8 is required for cell survival and patterning of the first branchial arch","volume":"13","author":"Trumpp","year":"1999","journal-title":"Genes Dev."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"dmm031526","DOI":"10.1242\/dmm.031526","article-title":"Increased FGF8 signaling promotes chondrogenic rather than osteogenic development in the embryonic skull","volume":"11","author":"Schmidt","year":"2018","journal-title":"Dis. Model. Mech."},{"key":"ref_35","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":"ref_36","doi-asserted-by":"crossref","first-page":"4","DOI":"10.1016\/j.ydbio.2016.07.001","article-title":"A novel ciliopathic skull defect arising from excess neural crest","volume":"417","author":"Tabler","year":"2016","journal-title":"Dev. Biol."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"1304","DOI":"10.1016\/S0960-9822(00)80052-4","article-title":"Inhibition of Sonic hedgehog signaling in vivo results in craniofacial neural crest cell death","volume":"9","author":"Ahlgren","year":"1999","journal-title":"Curr. Biol."},{"key":"ref_38","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":"ref_39","doi-asserted-by":"crossref","first-page":"376","DOI":"10.1002\/dvdy.23717","article-title":"Notch pathway regulation of neural crest cell development in vivo","volume":"241","author":"Mead","year":"2012","journal-title":"Dev. Dyn."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"1461","DOI":"10.1242\/dev.042911","article-title":"RBPjkappa-dependent Notch signaling regulates mesenchymal progenitor cell proliferation and differentiation during skeletal development","volume":"137","author":"Dong","year":"2010","journal-title":"Development"},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"530","DOI":"10.1101\/gad.317405","article-title":"Inactivation of TGFbeta signaling in neural crest stem cells leads to multiple defects reminiscent of DiGeorge syndrome","volume":"19","author":"Wurdak","year":"2005","journal-title":"Genes Dev."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"232","DOI":"10.4161\/cam.5.3.15498","article-title":"TGFbeta superfamily signaling in the neural crest lineage","volume":"5","author":"Conway","year":"2011","journal-title":"Cell Adh. Migr."},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1242\/dev.02200","article-title":"TGFbeta-mediated FGF signaling is crucial for regulating cranial neural crest cell proliferation during frontal bone development","volume":"133","author":"Sasaki","year":"2006","journal-title":"Development"},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"5269","DOI":"10.1242\/dev.00708","article-title":"Conditional inactivation of Tgfbr2 in cranial neural crest causes cleft palate and calvaria defects","volume":"130","author":"Ito","year":"2003","journal-title":"Development"},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"13467","DOI":"10.1074\/jbc.M112.431775","article-title":"TGF-beta-activated kinase 1 (Tak1) mediates agonist-induced Smad activation and linker region phosphorylation in embryonic craniofacial neural crest-derived cells","volume":"288","author":"Yumoto","year":"2013","journal-title":"J. Biol. Chem."},{"key":"ref_46","doi-asserted-by":"crossref","first-page":"1555","DOI":"10.1242\/dev.121590","article-title":"Animal models for studying neural crest development: Is the mouse different?","volume":"142","author":"Barriga","year":"2015","journal-title":"Development"},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"1438","DOI":"10.1172\/JCI38019","article-title":"Epithelial-mesenchymal transitions: The importance of changing cell state in development and disease","volume":"119","author":"Acloque","year":"2009","journal-title":"J. Clin. Investig."},{"key":"ref_48","doi-asserted-by":"crossref","first-page":"957","DOI":"10.1038\/nature07441","article-title":"Contact inhibition of locomotion in vivo controls neural crest directional migration","volume":"456","author":"Matthews","year":"2008","journal-title":"Nature"},{"key":"ref_49","doi-asserted-by":"crossref","first-page":"3404","DOI":"10.1002\/dvdy.21758","article-title":"Wnt11ris required for cranial neural crest migration","volume":"237","author":"Matthews","year":"2008","journal-title":"Dev. Dyn."},{"key":"ref_50","doi-asserted-by":"crossref","first-page":"3287","DOI":"10.1242\/dev.067306","article-title":"A novel role for MuSK and non-canonical Wnt signaling during segmental neural crest cell migration","volume":"138","author":"Banerjee","year":"2011","journal-title":"Development"},{"key":"ref_51","doi-asserted-by":"crossref","first-page":"2587","DOI":"10.1242\/dev.01857","article-title":"Essential role of non-canonical Wnt signalling in neural crest migration","volume":"132","author":"Araya","year":"2005","journal-title":"Development"},{"key":"ref_52","doi-asserted-by":"crossref","first-page":"3153","DOI":"10.1242\/dev.111427","article-title":"Vangl-dependent planar cell polarity signalling is not required for neural crest migration in mammals","volume":"141","author":"Pryor","year":"2014","journal-title":"Development"},{"key":"ref_53","doi-asserted-by":"crossref","first-page":"e23276","DOI":"10.1002\/dvg.23276","article-title":"Specification and formation of the neural crest: Perspectives on lineage segregation","volume":"57","author":"Prasad","year":"2019","journal-title":"Genesis"},{"key":"ref_54","doi-asserted-by":"crossref","first-page":"S110","DOI":"10.1016\/j.ydbio.2018.05.013","article-title":"Neurocristopathies: New insights 150 years after the neural crest discovery","volume":"444","author":"Cerrizuela","year":"2018","journal-title":"Dev. Biol."},{"key":"ref_55","doi-asserted-by":"crossref","first-page":"215","DOI":"10.1016\/S0022-3476(64)80522-9","article-title":"Mandibulofacial dysostosis, a familial study of five generations","volume":"65","author":"Rovin","year":"1964","journal-title":"J. Pediatr."},{"key":"ref_56","first-page":"190","article-title":"Cases with symmetrical congenital notches in the outer part of each lower lid and defective development of the malar bones","volume":"20","author":"Collins","year":"1900","journal-title":"Trans. Ophthalmol. Soc. UK"},{"key":"ref_57","first-page":"143","article-title":"The mandibulofacial dysostosis; a new hereditary syndrome","volume":"27","author":"Franceschetti","year":"1949","journal-title":"Acta Ophthalmol."},{"key":"ref_58","doi-asserted-by":"crossref","first-page":"275","DOI":"10.1038\/ejhg.2008.221","article-title":"Treacher Collins syndrome: Etiology, pathogenesis and prevention","volume":"17","author":"Trainor","year":"2009","journal-title":"Eur. J. Hum. Genet."},{"key":"ref_59","doi-asserted-by":"crossref","first-page":"523","DOI":"10.1038\/nature14978","article-title":"Cell-fate determination by ubiquitin-dependent regulation of translation","volume":"525","author":"Werner","year":"2015","journal-title":"Nature"},{"key":"ref_60","doi-asserted-by":"crossref","first-page":"266","DOI":"10.1002\/bdrb.20252","article-title":"The etiology of cleft palate: A 50-year search for mechanistic and molecular understanding","volume":"89","author":"Abbott","year":"2010","journal-title":"Birth Defects Res. Part B Dev. Reprod. Toxicol."},{"key":"ref_61","doi-asserted-by":"crossref","first-page":"67","DOI":"10.3389\/fphys.2016.00067","article-title":"Epidemiology, Etiology, and Treatment of Isolated Cleft Palate","volume":"7","author":"Burg","year":"2016","journal-title":"Front. Physiol."},{"key":"ref_62","doi-asserted-by":"crossref","first-page":"432","DOI":"10.1136\/bmj.39458.563611.AE","article-title":"Familial risk of oral clefts by morphological type and severity: Population based cohort study of first degree relatives","volume":"336","author":"Sivertsen","year":"2008","journal-title":"BMJ"},{"key":"ref_63","doi-asserted-by":"crossref","first-page":"313","DOI":"10.1097\/EDE.0b013e3182125f9c","article-title":"Risk of Oral Clefts in Twins","volume":"22","author":"Grosen","year":"2011","journal-title":"Epidemiology"},{"key":"ref_64","doi-asserted-by":"crossref","first-page":"231","DOI":"10.1242\/dev.067082","article-title":"Palatogenesis: Morphogenetic and molecular mechanisms of secondary palate development","volume":"139","author":"Bush","year":"2012","journal-title":"Development"},{"key":"ref_65","doi-asserted-by":"crossref","unstructured":"Jane\u010dkov\u00e1, E., Feng, J., Li, J., Rodriguez, G., and Chai, Y. (2019). Dynamic activation of Wnt, Fgf, and Hh signaling during soft palate development. PLoS ONE, 14.","DOI":"10.1371\/journal.pone.0223879"},{"key":"ref_66","doi-asserted-by":"crossref","first-page":"4709","DOI":"10.1242\/dev.099028","article-title":"Pax9 regulates a molecular network involving Bmp4, Fgf10, Shh signaling and the Osr2 transcription factor to control palate morphogenesis","volume":"140","author":"Zhou","year":"2013","journal-title":"Development"},{"key":"ref_67","doi-asserted-by":"crossref","first-page":"35","DOI":"10.4103\/2231-0746.186134","article-title":"Pierre Robin sequence: Subdivision, data, theories, and treatment\u2014Part 2: Syndromic and nonsyndromic Pierre Robin sequence","volume":"6","author":"Zwahlen","year":"2016","journal-title":"Ann. Maxillofac. Surg."},{"key":"ref_68","doi-asserted-by":"crossref","first-page":"31","DOI":"10.4103\/2231-0746.186133","article-title":"Pierre Robin sequence: Subdivision, data, theories, and treatment\u2014Part 1: History, subdivisions, and data","volume":"6","author":"Zwahlen","year":"2016","journal-title":"Ann. Maxillofac. Surg."},{"key":"ref_69","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1007\/s00441-018-2944-5","article-title":"Conditional deletion of Bmp2 in cranial neural crest cells recapitulates Pierre Robin sequence in mice","volume":"376","author":"Chen","year":"2018","journal-title":"Cell Tissue Res."},{"key":"ref_70","doi-asserted-by":"crossref","first-page":"197","DOI":"10.1016\/j.cps.2018.11.005","article-title":"Gilardino, Treacher Collins Syndrome","volume":"46","author":"Aljerian","year":"2019","journal-title":"Clin. Plast. Surg."},{"key":"ref_71","doi-asserted-by":"crossref","first-page":"44","DOI":"10.4103\/2231-0746.186136","article-title":"Pierre Robin sequence: Subdivision, data, theories, and treatment\u2014Part 4: Recommended management and treatment of Pierre Robin sequence and its application","volume":"6","author":"Zwahlen","year":"2016","journal-title":"Ann. Maxillofac. Surg."},{"key":"ref_72","first-page":"149","article-title":"Priorities for public health research on craniosynostosis: Summary and recommendations from a Centers for Disease Control and Prevention-sponsored meeting","volume":"146","author":"Rasmussen","year":"2007","journal-title":"Am. J. Med. Genet. Part A"},{"key":"ref_73","doi-asserted-by":"crossref","first-page":"1252","DOI":"10.1097\/SCS.0b013e3181acdf9a","article-title":"Intellectual Outcomes Following Protocol Management in Crouzon, Pfeiffer, and Muenke Syndromes","volume":"20","author":"Flapper","year":"2009","journal-title":"J. Craniofacial Surg."},{"key":"ref_74","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1597\/11-138","article-title":"Parameters of care for craniosynostosis","volume":"49","author":"McCarthy","year":"2012","journal-title":"Cleft Palate Craniofac. J."},{"key":"ref_75","doi-asserted-by":"crossref","first-page":"160","DOI":"10.1002\/(SICI)1096-8628(19990716)85:2<160::AID-AJMG11>3.0.CO;2-R","article-title":"Ocular anterior chamber dysgenesis in craniosynostosis syndromes with a fibroblast growth factor receptor 2 mutation","volume":"85","author":"Okajima","year":"1999","journal-title":"Am. J. Med. Genet."},{"key":"ref_76","doi-asserted-by":"crossref","first-page":"40","DOI":"10.3171\/2011.4.PEDS1160","article-title":"Treatment for delayed presentation of sagittal synostosis: Challenges pertaining to occult intracranial hypertension","volume":"8","author":"Seruya","year":"2011","journal-title":"J. Neurosurg. Pediatr."},{"key":"ref_77","doi-asserted-by":"crossref","first-page":"581","DOI":"10.1002\/ajmg.1320470507","article-title":"Sutural biology and the correlates of craniosynostosis","volume":"47","author":"Cohen","year":"1993","journal-title":"Am. J. Med. Genet."},{"key":"ref_78","doi-asserted-by":"crossref","first-page":"53","DOI":"10.1007\/BF00706732","article-title":"Functional prognosis of surgical treatment of craniosynostosis","volume":"1","author":"Abe","year":"1985","journal-title":"Childs Nerv. Syst."},{"key":"ref_79","doi-asserted-by":"crossref","first-page":"411","DOI":"10.1007\/s00381-011-1615-6","article-title":"Craniofacial reconstruction as a treatment for elevated intracranial pressure","volume":"28","author":"Baird","year":"2011","journal-title":"Childs Nerv. Syst."},{"key":"ref_80","doi-asserted-by":"crossref","first-page":"645","DOI":"10.1007\/s003810000320","article-title":"Management of craniosynostoses","volume":"16","author":"Renier","year":"2000","journal-title":"Childs Nerv. Syst."},{"key":"ref_81","doi-asserted-by":"crossref","unstructured":"Torrance, J.S., Wincenciak, J., Hahn, A.C., Debruine, L.M., and Jones, B.C. (2014). The Relative Contributions of Facial Shape and Surface Information to Perceptions of Attractiveness and Dominance. PLoS ONE, 9.","DOI":"10.1371\/journal.pone.0104415"},{"key":"ref_82","doi-asserted-by":"crossref","first-page":"053","DOI":"10.1055\/s-0032-1320063","article-title":"Nonsyndromic Craniosynostosis","volume":"26","author":"Garza","year":"2012","journal-title":"Semin. Plast. Surg."},{"key":"ref_83","doi-asserted-by":"crossref","first-page":"e391","DOI":"10.1542\/peds.2009-3491","article-title":"Prevalence and complications of single-gene and chromosomal disorders in craniosynostosis","volume":"126","author":"Wilkie","year":"2010","journal-title":"Pediatrics"},{"key":"ref_84","first-page":"223","article-title":"Analysis of the Fgfr2C342Y mouse model shows condensation defects due to misregulation of Sox9 expression in prechondrocytic mesenchyme","volume":"6","author":"Peskett","year":"2017","journal-title":"Biol. Open"},{"key":"ref_85","doi-asserted-by":"crossref","first-page":"855","DOI":"10.1242\/dev.028605","article-title":"EphA4 as an effector of Twist1 in the guidance of osteogenic precursor cells during calvarial bone growth and in craniosynostosis","volume":"136","author":"Ting","year":"2009","journal-title":"Development"},{"key":"ref_86","doi-asserted-by":"crossref","first-page":"dmm035311","DOI":"10.1242\/dmm.035311","article-title":"Overexpression of Fgfr2c causes craniofacial bone hypoplasia and ameliorates craniosynostosis in the Crouzon mouse","volume":"11","author":"Lee","year":"2018","journal-title":"Dis. Model. Mech."},{"key":"ref_87","doi-asserted-by":"crossref","first-page":"451","DOI":"10.1007\/s00223-013-9701-2","article-title":"Further analysis of the Crouzon mouse: Effects of the FGFR2(C342Y) mutation are cranial bone-dependent","volume":"92","author":"Liu","year":"2013","journal-title":"Calcif. Tissue Int."},{"key":"ref_88","doi-asserted-by":"crossref","first-page":"242","DOI":"10.1016\/j.ydbio.2016.03.026","article-title":"FGFR2c-mediated ERK-MAPK activity regulates coronal suture development","volume":"415","author":"Pfaff","year":"2016","journal-title":"Dev. Biol."},{"key":"ref_89","doi-asserted-by":"crossref","first-page":"292","DOI":"10.1080\/03008200390181799","article-title":"Activating (P253R, C278F) and dominant negative mutations of FGFR2: Differential effects on calvarial bone cell proliferation, differentiation, and mineralization","volume":"44","author":"Ratisoontorn","year":"2003","journal-title":"Connect. Tissue Res."},{"key":"ref_90","doi-asserted-by":"crossref","first-page":"258","DOI":"10.1016\/j.ydbio.2010.08.010","article-title":"Jagged1 functions downstream of Twist1 in the specification of the coronal suture and the formation of a boundary between osteogenic and non-osteogenic cells","volume":"347","author":"Yen","year":"2010","journal-title":"Dev. Biol."},{"key":"ref_91","doi-asserted-by":"crossref","first-page":"273","DOI":"10.1016\/j.ydbio.2009.01.026","article-title":"Early onset of craniosynostosis in an Apert mouse model reveals critical features of this pathology","volume":"328","author":"Holmes","year":"2009","journal-title":"Dev. Biol."},{"key":"ref_92","doi-asserted-by":"crossref","first-page":"169","DOI":"10.1016\/S8756-3282(03)00222-9","article-title":"A Ser252Trp [corrected] substitution in mouse fibroblast growth factor receptor 2 (Fgfr2) results in craniosynostosis","volume":"33","author":"Chen","year":"2003","journal-title":"Bone"},{"key":"ref_93","doi-asserted-by":"crossref","first-page":"283","DOI":"10.1016\/j.ydbio.2012.05.026","article-title":"Mesodermal expression of Fgfr2S252W is necessary and sufficient to induce craniosynostosis in a mouse model of Apert syndrome","volume":"368","author":"Holmes","year":"2012","journal-title":"Dev. Biol."},{"key":"ref_94","first-page":"4026","article-title":"Dysregulated PDGFRalpha signaling alters coronal suture morphogenesis and leads to craniosynostosis through endochondral ossification","volume":"144","author":"He","year":"2017","journal-title":"Development"},{"key":"ref_95","doi-asserted-by":"crossref","first-page":"806","DOI":"10.1038\/nrn1221","article-title":"Cranial neural crest and the building of the vertebrate head","volume":"4","author":"Santagati","year":"2003","journal-title":"Nat. Rev. Neurosci."},{"key":"ref_96","doi-asserted-by":"crossref","first-page":"106","DOI":"10.1006\/dbio.2001.0487","article-title":"Tissue Origins and Interactions in the Mammalian Skull Vault","volume":"241","author":"Jiang","year":"2002","journal-title":"Dev. Biol."},{"key":"ref_97","doi-asserted-by":"crossref","first-page":"797","DOI":"10.1016\/j.mod.2008.06.007","article-title":"Cell lineage in mammalian craniofacial mesenchyme","volume":"125","author":"Yoshida","year":"2008","journal-title":"Mech. Dev."},{"key":"ref_98","doi-asserted-by":"crossref","first-page":"e23248","DOI":"10.1002\/dvg.23248","article-title":"A tale of two cities: The genetic mechanisms governing calvarial bone development","volume":"57","author":"Ferguson","year":"2018","journal-title":"Genesis"},{"key":"ref_99","doi-asserted-by":"crossref","first-page":"9360","DOI":"10.1073\/pnas.1631288100","article-title":"Sox9 is required for determination of the chondrogenic cell lineage in the cranial neural crest","volume":"100","author":"Akiyama","year":"2003","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_100","doi-asserted-by":"crossref","first-page":"689","DOI":"10.1002\/stem.607","article-title":"Transforming Growth Factor \u03b2-Mediated Sox10 Suppression Controls Mesenchymal Progenitor Generation in Neural Crest Stem Cells","volume":"29","author":"John","year":"2011","journal-title":"Stem Cells"},{"key":"ref_101","doi-asserted-by":"crossref","first-page":"867","DOI":"10.1242\/dev.094342","article-title":"Ezh2 is required for neural crest-derived cartilage and bone formation","volume":"141","author":"Schwarz","year":"2014","journal-title":"Development"},{"key":"ref_102","doi-asserted-by":"crossref","first-page":"138","DOI":"10.1002\/(SICI)1521-1878(200002)22:2<138::AID-BIES5>3.0.CO;2-4","article-title":"All for one and one for all: Condensations and the initiation of skeletal development","volume":"22","author":"Hall","year":"2000","journal-title":"Bioessays"},{"key":"ref_103","doi-asserted-by":"crossref","first-page":"6698","DOI":"10.1073\/pnas.111092198","article-title":"Haploinsufficiency of Sox9 results in defective cartilage primordia and premature skeletal mineralization","volume":"98","author":"Bi","year":"2001","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_104","doi-asserted-by":"crossref","first-page":"28","DOI":"10.1016\/j.ydbio.2010.04.007","article-title":"Inactivation of Msx1 and Msx2 in neural crest reveals an unexpected role in suppressing heterotopic bone formation in the head","volume":"343","author":"Roybal","year":"2010","journal-title":"Dev. Biol."},{"key":"ref_105","doi-asserted-by":"crossref","first-page":"335","DOI":"10.1016\/j.ydbio.2007.08.028","article-title":"Cell fate specification during calvarial bone and suture development","volume":"311","author":"Rice","year":"2007","journal-title":"Dev. Biol."},{"key":"ref_106","doi-asserted-by":"crossref","first-page":"729","DOI":"10.1016\/j.mod.2007.06.006","article-title":"Concerted action of Msx1 and Msx2 in regulating cranial neural crest cell differentiation during frontal bone development","volume":"124","author":"Han","year":"2007","journal-title":"Mech. Dev."},{"key":"ref_107","doi-asserted-by":"crossref","first-page":"6131","DOI":"10.1242\/dev.00793","article-title":"Msx2 and Twist cooperatively control the development of the neural crest-derived skeletogenic mesenchyme of the murine skull vault","volume":"130","author":"Ishii","year":"2003","journal-title":"Development"},{"key":"ref_108","doi-asserted-by":"crossref","first-page":"765","DOI":"10.1016\/S0092-8674(00)80259-7","article-title":"Cbfa1, a candidate gene for cleidocranial dysplasia syndrome, is essential for osteoblast differentiation and bone development","volume":"89","author":"Otto","year":"1997","journal-title":"Cell"},{"key":"ref_109","doi-asserted-by":"crossref","first-page":"673","DOI":"10.1016\/j.bone.2011.07.023","article-title":"Early onset of Runx2 expression caused craniosynostosis, ectopic bone formation, and limb defects","volume":"49","author":"Maeno","year":"2011","journal-title":"Bone"},{"key":"ref_110","doi-asserted-by":"crossref","first-page":"755","DOI":"10.1016\/S0092-8674(00)80258-5","article-title":"Targeted Disruption of Cbfa1 Results in a Complete Lack of Bone Formation owing to Maturational Arrest of Osteoblasts","volume":"89","author":"Komori","year":"1997","journal-title":"Cell"},{"key":"ref_111","doi-asserted-by":"crossref","first-page":"17","DOI":"10.1016\/S0092-8674(01)00622-5","article-title":"The Novel Zinc Finger-Containing Transcription Factor Osterix Is Required for Osteoblast Differentiation and Bone Formation","volume":"108","author":"Nakashima","year":"2002","journal-title":"Cell"},{"key":"ref_112","doi-asserted-by":"crossref","first-page":"1845","DOI":"10.1242\/dev.127.9.1845","article-title":"Integration of FGF and TWIST in calvarial bone and suture development","volume":"127","author":"Rice","year":"2000","journal-title":"Development"},{"key":"ref_113","doi-asserted-by":"crossref","first-page":"265","DOI":"10.1046\/j.0909-8836.1999.eos107406.x","article-title":"Apoptosis in murine calvarial bone and suture development","volume":"107","author":"Rice","year":"1999","journal-title":"Eur. J. Oral Sci."},{"key":"ref_114","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1016\/j.ydbio.2008.03.037","article-title":"Twist1 homodimers enhance FGF responsiveness of the cranial sutures and promote suture closure","volume":"318","author":"Connerney","year":"2008","journal-title":"Dev. Biol."},{"key":"ref_115","doi-asserted-by":"crossref","first-page":"686","DOI":"10.1101\/gad.9.6.686","article-title":"Twist is required in head mesenchyme for cranial neural tube morphogenesis","volume":"9","author":"Chen","year":"1995","journal-title":"Genes Dev."},{"key":"ref_116","doi-asserted-by":"crossref","first-page":"251","DOI":"10.1006\/dbio.2002.0699","article-title":"Twist Function Is Required for the Morphogenesis of the Cephalic Neural Tube and the Differentiation of the Cranial Neural Crest Cells in the Mouse Embryo","volume":"247","author":"Soo","year":"2002","journal-title":"Dev. Biol."},{"key":"ref_117","doi-asserted-by":"crossref","first-page":"144","DOI":"10.1002\/dvdy.24367","article-title":"Twist1contributes to cranial bone initiation and dermal condensation by maintaining wnt signaling responsiveness","volume":"245","author":"Goodnough","year":"2015","journal-title":"Dev. Dyn."},{"key":"ref_118","doi-asserted-by":"crossref","first-page":"437","DOI":"10.1111\/j.1469-7580.2005.00411.x","article-title":"Twist is required for establishment of the mouse coronal suture","volume":"206","author":"Yoshida","year":"2005","journal-title":"J. Anat."},{"key":"ref_119","doi-asserted-by":"crossref","first-page":"3375","DOI":"10.1242\/dev.124.17.3375","article-title":"Fgfr2 and osteopontin domains in the developing skull vault are mutually exclusive and can be altered by locally applied FGF2","volume":"124","author":"Iseki","year":"1997","journal-title":"Development"},{"key":"ref_120","doi-asserted-by":"crossref","first-page":"5611","DOI":"10.1242\/dev.126.24.5611","article-title":"Fgfr1 and Fgfr2 have distinct differentiation- and proliferation-related roles in the developing mouse skull vault","volume":"126","author":"Iseki","year":"1999","journal-title":"Development"},{"key":"ref_121","doi-asserted-by":"crossref","first-page":"158","DOI":"10.1159\/000202789","article-title":"Differential FGF ligands and FGF receptors expression pattern in frontal and parietal calvarial bones","volume":"190","author":"Quarto","year":"2009","journal-title":"Cells Tissues Organs"},{"key":"ref_122","doi-asserted-by":"crossref","first-page":"48","DOI":"10.1159\/000493106","article-title":"The Osteogenic Potential of the Neural Crest Lineage May Contribute to Craniosynostosis","volume":"10","author":"Doro","year":"2018","journal-title":"Mol. Syndr."},{"key":"ref_123","doi-asserted-by":"crossref","first-page":"269","DOI":"10.1038\/ng1194-269","article-title":"A common mutation in the fibroblast growth factor receptor 1 gene in Pfeiffer syndrome","volume":"8","author":"Muenke","year":"1994","journal-title":"Nat. Genet."},{"key":"ref_124","doi-asserted-by":"crossref","first-page":"2001","DOI":"10.1093\/hmg\/9.13.2001","article-title":"A Pro250Arg substitution in mouse Fgfr1 causes increased expression of Cbfa1 and premature fusion of calvarial sutures","volume":"9","author":"Zhou","year":"2000","journal-title":"Hum. Mol. Genet."},{"key":"ref_125","doi-asserted-by":"crossref","first-page":"22","DOI":"10.1002\/1096-8628(20000703)93:1<22::AID-AJMG5>3.0.CO;2-U","article-title":"Clinical findings in a patient with FGFR1 P252R mutation and comparison with the literature","volume":"93","author":"Roscioli","year":"2000","journal-title":"Am. J. Med. Genet."},{"key":"ref_126","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1038\/ng0295-165","article-title":"Apert syndrome results from localized mutations of FGFR2 and is allelic with Crouzon syndrome","volume":"9","author":"Wilkie","year":"1995","journal-title":"Nat. Genet."},{"key":"ref_127","doi-asserted-by":"crossref","first-page":"1229","DOI":"10.1093\/hmg\/4.7.1229","article-title":"Novel FGFR2 mutations in Crouzon and Jackson-Weiss syndromes show allelic heterogeneity and phenotypic variability","volume":"4","author":"Park","year":"1995","journal-title":"Hum. Mol. Genet."},{"key":"ref_128","doi-asserted-by":"crossref","first-page":"275","DOI":"10.1038\/ng1194-275","article-title":"Jackson-Weiss and Crouzon syndromes are allelic with mutations in fibroblast growth factor receptor 2","volume":"8","author":"Jabs","year":"1994","journal-title":"Nat. Genet."},{"key":"ref_129","doi-asserted-by":"crossref","first-page":"1505","DOI":"10.1007\/s00381-012-1872-z","article-title":"Mouse models of Apert syndrome","volume":"28","author":"Holmes","year":"2012","journal-title":"Childs Nerv. Syst."},{"key":"ref_130","doi-asserted-by":"crossref","first-page":"12555","DOI":"10.1073\/pnas.0405031101","article-title":"A gain-of-function mutation of Fgfr2c demonstrates the roles of this receptor variant in osteogenesis","volume":"101","author":"Eswarakumar","year":"2004","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_131","doi-asserted-by":"crossref","first-page":"369","DOI":"10.1038\/ejhg.2010.235","article-title":"Craniosynostosis","volume":"19","author":"Johnson","year":"2011","journal-title":"Eur. J. Hum. Genet."},{"key":"ref_132","first-page":"555","article-title":"A unique point mutation in the fibroblast growth factor receptor 3 gene (FGFR3) defines a new craniosynostosis syndrome","volume":"60","author":"Muenke","year":"1997","journal-title":"Am. J. Hum. Genet."},{"key":"ref_133","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1002\/dvdy.21790","article-title":"Skeletal analysis of the Fgfr3P244R mouse, a genetic model for the Muenke craniosynostosis syndrome","volume":"238","author":"Twigg","year":"2008","journal-title":"Dev. Dyn."},{"key":"ref_134","doi-asserted-by":"crossref","first-page":"42","DOI":"10.1038\/ng0197-42","article-title":"Mutations of the TWIST gene in the Saethre-Chotzene syndrome","volume":"15","author":"Lajeunie","year":"1997","journal-title":"Nat. Genet."},{"key":"ref_135","first-page":"90","article-title":"Craniosynostosis inTwist heterozygous mice: A model for Saethre-Chotzen syndrome","volume":"268","author":"Carver","year":"2002","journal-title":"Anat. Rec. Adv. Integr. Anat. Evol. Biol."},{"key":"ref_136","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1016\/0030-4220(88)90358-1","article-title":"Craniofrontonasal dysplasia: Phenotypic expression in females and males and genetic considerations","volume":"65","author":"Grutzner","year":"1988","journal-title":"Oral Surgery Oral Med. Oral Pathol."},{"key":"ref_137","doi-asserted-by":"crossref","first-page":"8652","DOI":"10.1073\/pnas.0402819101","article-title":"Mutations of ephrin-B1 (EFNB1), a marker of tissue boundary formation, cause craniofrontonasal syndrome","volume":"101","author":"Twigg","year":"2004","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_138","doi-asserted-by":"crossref","first-page":"443","DOI":"10.1016\/0092-8674(93)90379-5","article-title":"A mutation in the homeodomain of the human MSX2 gene in a family affected with autosomal dominant craniosynostosis","volume":"75","author":"Jabs","year":"1993","journal-title":"Cell"},{"key":"ref_139","doi-asserted-by":"crossref","first-page":"444","DOI":"10.1002\/ajmg.1320460420","article-title":"Newly recognized autosomal dominant disorder with craniosynostosis","volume":"46","author":"Warman","year":"1993","journal-title":"Am. J. Med. Genet."},{"key":"ref_140","doi-asserted-by":"crossref","first-page":"1915","DOI":"10.1093\/hmg\/5.12.1915","article-title":"The molecular basis of Boston-type craniosynostosis: The Pro148-->His mutation in the N-terminal arm of the MSX2 homeodomain stabilizes DNA binding without altering nucleotide sequence preferences","volume":"5","author":"Ma","year":"1996","journal-title":"Hum. Mol. Genet."},{"key":"ref_141","doi-asserted-by":"crossref","first-page":"295","DOI":"10.1016\/j.ydbio.2012.12.004","article-title":"The mesenchymal architecture of the cranial mesoderm of mouse embryos is disrupted by the loss of Twist1 function","volume":"374","author":"Bildsoe","year":"2012","journal-title":"Dev. Biol."},{"key":"ref_142","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1016\/S1534-5807(03)00198-9","article-title":"Control of Skeletal Patterning by EphrinB1-EphB Interactions","volume":"5","author":"Compagni","year":"2003","journal-title":"Dev. Cell"},{"key":"ref_143","doi-asserted-by":"crossref","first-page":"2320","DOI":"10.1101\/gad.228957.113","article-title":"Genetic rescue of Muenke syndrome model hearing loss reveals prolonged FGF-dependent plasticity in cochlear supporting cell fates","volume":"27","author":"Mansour","year":"2013","journal-title":"Genes Dev."},{"key":"ref_144","doi-asserted-by":"crossref","first-page":"1483","DOI":"10.1007\/s00381-012-1778-9","article-title":"Phenotype profile of a genetic mouse model for Muenke syndrome","volume":"28","author":"Nah","year":"2012","journal-title":"Childs Nerv. Syst."},{"key":"ref_145","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1016\/j.ajhg.2012.02.005","article-title":"Bent Bone Dysplasia-FGFR2 type, a Distinct Skeletal Disorder, Has Deficient Canonical FGF Signaling","volume":"90","author":"Merrill","year":"2012","journal-title":"Am. J. Hum. Genet."},{"key":"ref_146","doi-asserted-by":"crossref","first-page":"5659","DOI":"10.1093\/hmg\/ddu282","article-title":"Bent bone dysplasia syndrome reveals nucleolar activity for FGFR2 in ribosomal DNA transcription","volume":"23","author":"Neben","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"ref_147","doi-asserted-by":"crossref","first-page":"7","DOI":"10.7554\/eLife.37024","article-title":"Altered bone growth dynamics prefigure craniosynostosis in a zebrafish model of Saethre-Chotzen syndrome","volume":"7","author":"Teng","year":"2018","journal-title":"eLife"},{"key":"ref_148","doi-asserted-by":"crossref","first-page":"304","DOI":"10.1038\/ng.2531","article-title":"Mutations in TCF12, encoding a basic helix-loop-helix partner of TWIST1, are a frequent cause of coronal craniosynostosis","volume":"45","author":"Sharma","year":"2013","journal-title":"Nat. Genet."},{"key":"ref_149","doi-asserted-by":"crossref","first-page":"81","DOI":"10.1016\/j.bone.2014.06.040","article-title":"Tissue-nonspecific alkaline phosphatase deficiency causes abnormal craniofacial bone development in the Alpl(\u2212\/\u2212) mouse model of infantile hypophosphatasia","volume":"67","author":"Liu","year":"2014","journal-title":"Bone"},{"key":"ref_150","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1016\/j.bone.2018.10.013","article-title":"Tissue nonspecific alkaline phosphatase promotes calvarial progenitor cell cycle progression and cytokinesis via Erk1,2","volume":"120","author":"Nam","year":"2019","journal-title":"Bone"},{"key":"ref_151","doi-asserted-by":"crossref","unstructured":"Li, S., Quarto, N., and Longaker, M.T. (2010). Activation of FGF Signaling Mediates Proliferative and Osteogenic Differences between Neural Crest Derived Frontal and Mesoderm Parietal Derived Bone. PLoS ONE, 5.","DOI":"10.1371\/journal.pone.0014033"},{"key":"ref_152","doi-asserted-by":"crossref","first-page":"495","DOI":"10.1387\/ijdb.170051gc","article-title":"Contribution of cranial neural crest cells to mouse skull development","volume":"61","author":"Wu","year":"2017","journal-title":"Int. J. Dev. Biol."},{"key":"ref_153","doi-asserted-by":"crossref","first-page":"1680","DOI":"10.1359\/jbmr.091116","article-title":"Origin Matters: Differences in Embryonic Tissue Origin and Wnt Signaling Determine the Osteogenic Potential and Healing Capacity of Frontal and Parietal Calvarial Bones","volume":"25","author":"Quarto","year":"2009","journal-title":"J. Bone Miner. Res."},{"key":"ref_154","doi-asserted-by":"crossref","first-page":"8","DOI":"10.7554\/eLife.52814","article-title":"Resolving homology in the face of shifting germ layer origins: Lessons from a major skull vault boundary","volume":"8","author":"Teng","year":"2019","journal-title":"eLife"},{"key":"ref_155","doi-asserted-by":"crossref","first-page":"12","DOI":"10.1111\/j.1399-0004.1992.tb03620.x","article-title":"Birth prevalence studies of the Crouzon syndrome: Comparison of direct and indirect methods","volume":"41","author":"Cohen","year":"2008","journal-title":"Clin. Genet."},{"key":"ref_156","doi-asserted-by":"crossref","first-page":"26037","DOI":"10.1074\/jbc.270.44.26037","article-title":"Constitutive Activation of Fibroblast Growth Factor Receptor-2 by a Point Mutation Associated with Crouzon Syndrome","volume":"270","author":"Neilson","year":"1995","journal-title":"J. Biol. Chem."},{"key":"ref_157","doi-asserted-by":"crossref","first-page":"7894","DOI":"10.1073\/pnas.93.15.7894","article-title":"Constitutive receptor activation by Crouzon syndrome mutations in fibroblast growth factor receptor (FGFR)2 and FGFR2\/Neu chimeras","volume":"93","author":"Galvin","year":"1996","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_158","doi-asserted-by":"crossref","first-page":"4567","DOI":"10.1073\/pnas.95.8.4567","article-title":"Activating mutations in the extracellular domain of the fibroblast growth factor receptor 2 function by disruption of the disulfide bond in the third immunoglobulin-like domain","volume":"95","author":"Robertson","year":"1998","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_159","doi-asserted-by":"crossref","first-page":"98","DOI":"10.1038\/ng0994-98","article-title":"Mutations in the fibroblast growth factor receptor 2 gene cause Crouzon syndrome","volume":"8","author":"Reardon","year":"1994","journal-title":"Nat. Genet."},{"key":"ref_160","doi-asserted-by":"crossref","first-page":"1297","DOI":"10.1083\/jcb.149.6.1297","article-title":"Signaling by Fibroblast Growth Factors (Fgf) and Fibroblast Growth Factor Receptor 2 (Fgfr2)\u2013Activating Mutations Blocks Mineralization and Induces Apoptosis in Osteoblasts","volume":"149","author":"Mansukhani","year":"2000","journal-title":"J. Cell Biol."},{"key":"ref_161","doi-asserted-by":"crossref","first-page":"758","DOI":"10.1002\/ajmg.1320450618","article-title":"Visceral anomalies in the Apert syndrome","volume":"45","author":"Cohen","year":"1993","journal-title":"Am. J. Med. Genet."},{"key":"ref_162","doi-asserted-by":"crossref","first-page":"624","DOI":"10.1002\/ajmg.1320470509","article-title":"Skeletal abnormalities in the Apert syndrome","volume":"47","author":"Cohen","year":"1993","journal-title":"Am. J. Med. Genet."},{"key":"ref_163","doi-asserted-by":"crossref","unstructured":"Chen, P., Zhang, L., Weng, T., Zhang, S., Sun, S., Chang, M., Li, Y., Zhang, B., and Zhang, L. (2014). A Ser252Trp Mutation in Fibroblast Growth Factor Receptor 2 (FGFR2) Mimicking Human Apert Syndrome Reveals an Essential Role for FGF Signaling in the Regulation of Endochondral Bone Formation. PLoS ONE, 9.","DOI":"10.1371\/journal.pone.0087311"},{"key":"ref_164","doi-asserted-by":"crossref","first-page":"1475","DOI":"10.1093\/hmg\/7.9.1475","article-title":"Apert syndrome mutations in fibroblast growth factor receptor 2 exhibit increased affinity for FGF ligand","volume":"7","author":"Anderson","year":"1998","journal-title":"Hum. Mol. Genet."},{"key":"ref_165","doi-asserted-by":"crossref","first-page":"14536","DOI":"10.1073\/pnas.97.26.14536","article-title":"Loss of fibroblast growth factor receptor 2 ligand-binding specificity in Apert syndrome","volume":"97","author":"Yu","year":"2000","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_166","doi-asserted-by":"crossref","first-page":"1833","DOI":"10.1016\/S0002-9440(10)64139-9","article-title":"Increased Osteoblast Apoptosis in Apert Craniosynostosis","volume":"158","author":"Lemonnier","year":"2001","journal-title":"Am. J. Pathol."},{"key":"ref_167","doi-asserted-by":"crossref","first-page":"1032","DOI":"10.1016\/j.bone.2008.02.009","article-title":"FGFR2-Cbl interaction in lipid rafts triggers attenuation of PI3K\/Akt signaling and osteoblast survival","volume":"42","author":"Dufour","year":"2008","journal-title":"Bone"},{"key":"ref_168","first-page":"1310","article-title":"Increased calvaria cell differentiation and bone matrix formation induced by fibroblast growth factor receptor 2 mutations in Apert syndrome","volume":"101","author":"Lomri","year":"1998","journal-title":"J. Clin. Investig."},{"key":"ref_169","doi-asserted-by":"crossref","first-page":"63","DOI":"10.1242\/dev.02171","article-title":"The homeoprotein engrailed 1 has pleiotropic functions in calvarial intramembranous bone formation and remodeling","volume":"133","author":"Deckelbaum","year":"2006","journal-title":"Development"},{"key":"ref_170","doi-asserted-by":"crossref","first-page":"341","DOI":"10.1016\/S0925-4773(99)00278-6","article-title":"Expression patterns of Twist and Fgfr1, -2 and -3 in the developing mouse coronal suture suggest a key role for twist in suture initiation and biogenesis","volume":"91","author":"Johnson","year":"2000","journal-title":"Mech. Dev."},{"key":"ref_171","doi-asserted-by":"crossref","first-page":"e23271","DOI":"10.1002\/dvg.23271","article-title":"Cellular organization and boundary formation in craniofacial development","volume":"57","author":"Kindberg","year":"2019","journal-title":"Genesis"},{"key":"ref_172","doi-asserted-by":"crossref","first-page":"386","DOI":"10.1038\/ncb3139","article-title":"The suture provides a niche for mesenchymal stem cells of craniofacial bones","volume":"17","author":"Zhao","year":"2015","journal-title":"Nat. Cell Biol."},{"key":"ref_173","doi-asserted-by":"crossref","first-page":"10526","DOI":"10.1038\/ncomms10526","article-title":"Stem cells of the suture mesenchyme in craniofacial bone development, repair and regeneration","volume":"7","author":"Maruyama","year":"2016","journal-title":"Nat. Commun."},{"key":"ref_174","doi-asserted-by":"crossref","first-page":"139","DOI":"10.15252\/embr.201541809","article-title":"Histones and histone modifications in perinuclear chromatin anchoring: From yeast to man","volume":"17","author":"Harr","year":"2016","journal-title":"EMBO Rep."},{"key":"ref_175","doi-asserted-by":"crossref","first-page":"281","DOI":"10.1080\/15476286.2016.1189071","article-title":"Histone availability as a strategy to control gene expression","volume":"14","author":"Prado","year":"2016","journal-title":"RNA Biol."},{"key":"ref_176","doi-asserted-by":"crossref","first-page":"2893","DOI":"10.1101\/gad.1035902","article-title":"Histone methyltransferase activity associated with a human multiprotein complex containing the Enhancer of Zeste protein","volume":"16","author":"Kuzmichev","year":"2002","journal-title":"Genes Dev."},{"key":"ref_177","doi-asserted-by":"crossref","first-page":"1039","DOI":"10.1126\/science.1076997","article-title":"Role of Histone H3 Lysine 27 Methylation in Polycomb-Group Silencing","volume":"298","author":"Cao","year":"2002","journal-title":"Science"},{"key":"ref_178","doi-asserted-by":"crossref","first-page":"27604","DOI":"10.1074\/jbc.M115.672345","article-title":"Epigenetic Control of Skeletal Development by the Histone Methyltransferase Ezh2*","volume":"290","author":"Dudakovic","year":"2015","journal-title":"J. Biol. Chem."},{"key":"ref_179","doi-asserted-by":"crossref","first-page":"12894","DOI":"10.1074\/jbc.RA118.002983","article-title":"Enhancer of zeste homolog 2 (Ezh2) controls bone formation and cell cycle progression during osteogenesis in mice","volume":"293","author":"Dudakovic","year":"2018","journal-title":"J. Biol. Chem."},{"key":"ref_180","doi-asserted-by":"crossref","first-page":"19001","DOI":"10.1074\/jbc.RA118.003909","article-title":"Loss of histone methyltransferase Ezh2 stimulates an osteogenic transcriptional program in chondrocytes but does not affect cartilage development","volume":"293","author":"Camilleri","year":"2018","journal-title":"J. Biol. Chem."},{"key":"ref_181","doi-asserted-by":"crossref","first-page":"52","DOI":"10.1002\/jcp.24680","article-title":"Histone Deacetylase Inhibition Destabilizes the Multi-Potent State of Uncommitted Adipose-Derived Mesenchymal Stromal Cells","volume":"230","author":"Dudakovic","year":"2015","journal-title":"J. Cell. Physiol."},{"key":"ref_182","doi-asserted-by":"crossref","first-page":"499","DOI":"10.1038\/nature12753","article-title":"Topology of mammalian developmental enhancers and their regulatory landscapes","volume":"502","author":"Duboule","year":"2013","journal-title":"Nature"},{"key":"ref_183","doi-asserted-by":"crossref","first-page":"852","DOI":"10.15252\/msb.20156492","article-title":"Hierarchical folding and reorganization of chromosomes are linked to transcriptional changes in cellular differentiation","volume":"11","author":"Fraser","year":"2015","journal-title":"Mol. Syst. Biol."},{"key":"ref_184","doi-asserted-by":"crossref","first-page":"517","DOI":"10.1038\/s41588-018-0338-y","article-title":"EZH2 oncogenic mutations drive epigenetic, transcriptional, and structural changes within chromatin domains","volume":"51","author":"Donaldson","year":"2019","journal-title":"Nat. Genet."},{"key":"ref_185","doi-asserted-by":"crossref","first-page":"2972","DOI":"10.1002\/ajmg.a.36229","article-title":"Weaver syndrome and EZH2 mutations: Clarifying the clinical phenotype","volume":"161","author":"Murray","year":"2013","journal-title":"Am. J. Med. Genet. Part A"},{"key":"ref_186","doi-asserted-by":"crossref","first-page":"1470","DOI":"10.1210\/jc.2017-01948","article-title":"Ezh2 Mutations Found in the Weaver Overgrowth Syndrome Cause a Partial Loss of H3K27 Histone Methyltransferase Activity","volume":"103","author":"Lui","year":"2018","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_187","doi-asserted-by":"crossref","first-page":"1581","DOI":"10.1016\/j.celrep.2018.03.129","article-title":"High-Resolution Epigenomic Atlas of Human Embryonic Craniofacial Development","volume":"23","author":"Wilderman","year":"2018","journal-title":"Cell Rep."},{"key":"ref_188","doi-asserted-by":"crossref","first-page":"232","DOI":"10.1002\/ajmg.c.31377","article-title":"The ontology of craniofacial development and malformation for translational craniofacial research","volume":"163","author":"Brinkley","year":"2013","journal-title":"Am. J. Med. Genet. Part C Semin. Med. Genet."},{"key":"ref_189","doi-asserted-by":"crossref","first-page":"107871","DOI":"10.1016\/j.celrep.2020.107871","article-title":"Integrated Transcriptome and Network Analysis Reveals Spatiotemporal Dynamics of Calvarial Suturogenesis","volume":"32","author":"Holmes","year":"2020","journal-title":"Cell Rep."},{"key":"ref_190","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1159\/000490480","article-title":"Structural Genome Variations Related to Craniosynostosis","volume":"10","author":"Poot","year":"2018","journal-title":"Mol. Syndr."},{"key":"ref_191","unstructured":"(2020, July 31). Online Mendelian Inheritance in Man, OMIM\u00ae. Available online: https:\/\/www.omim.org\/about."},{"key":"ref_192","doi-asserted-by":"crossref","first-page":"2155","DOI":"10.1089\/ten.tea.2008.0433","article-title":"Comparative Evaluation of Nanofibrous Scaffolding for Bone Regeneration in Critical-Size Calvarial Defects","volume":"15","author":"Woo","year":"2009","journal-title":"Tissue Eng. Part A"},{"key":"ref_193","doi-asserted-by":"crossref","first-page":"546","DOI":"10.1016\/j.tibtech.2012.07.005","article-title":"Recent advances in bone tissue engineering scaffolds","volume":"30","author":"Bose","year":"2012","journal-title":"Trends Biotechnol."},{"key":"ref_194","doi-asserted-by":"crossref","first-page":"20379","DOI":"10.1073\/pnas.1218052109","article-title":"In vivo directed differentiation of pluripotent stem cells for skeletal regeneration","volume":"109","author":"Levi","year":"2012","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_195","doi-asserted-by":"crossref","first-page":"10376","DOI":"10.1038\/ncomms10376","article-title":"Cell-free 3D scaffold with two-stage delivery of miRNA-26a to regenerate critical-sized bone defects","volume":"7","author":"Zhang","year":"2016","journal-title":"Nat. Commun."},{"key":"ref_196","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1097\/SCS.0000000000003893","article-title":"The Role of Skeletal Stem Cells in the Reconstruction of Bone Defects","volume":"28","author":"Murphy","year":"2017","journal-title":"J. Craniofacial Surg."},{"key":"ref_197","doi-asserted-by":"crossref","first-page":"598","DOI":"10.1177\/0885328217734824","article-title":"Cryogel scaffolds from patient-specific 3D-printed molds for personalized tissue-engineered bone regeneration in pediatric cleft-craniofacial defects","volume":"32","author":"Hixon","year":"2017","journal-title":"J. Biomater. Appl."},{"key":"ref_198","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.actbio.2018.10.016","article-title":"Pore size directs bone marrow stromal cell fate and tissue regeneration in nanofibrous macroporous scaffolds by mediating vascularization","volume":"82","author":"Gupte","year":"2018","journal-title":"Acta Biomater."},{"key":"ref_199","doi-asserted-by":"crossref","first-page":"514","DOI":"10.1038\/s41586-018-0650-9","article-title":"Mechanoresponsive stem cells acquire neural crest fate in jaw regeneration","volume":"563","author":"Ransom","year":"2018","journal-title":"Nature"},{"key":"ref_200","doi-asserted-by":"crossref","first-page":"161","DOI":"10.3389\/fphys.2017.00161","article-title":"Tissue Nonspecific Alkaline Phosphatase (TNAP) Regulates Cranial Base Growth and Synchondrosis Maturation","volume":"8","author":"Nam","year":"2017","journal-title":"Front. Physiol."},{"key":"ref_201","doi-asserted-by":"crossref","first-page":"652","DOI":"10.1359\/jbmr.081213","article-title":"FGF2 Stimulation of the Pyrophosphate-Generating Enzyme, PC-1, in Pre-Osteoblast Cells Is Mediated by RUNX2","volume":"24","author":"Hatch","year":"2008","journal-title":"J. Bone Miner. Res."},{"key":"ref_202","first-page":"292506","article-title":"Craniosynostosis-associated Fgfr2(C342Y) mutant bone marrow stromal cells exhibit cell autonomous abnormalities in osteoblast differentiation and bone formation","volume":"2013","author":"Liu","year":"2013","journal-title":"BioMed. Res. Int."},{"key":"ref_203","doi-asserted-by":"crossref","unstructured":"Nam, H.K., Vesel\u00e1, I., Schutte, S.D., and Hatch, N.E. (2020). Viral delivery of tissue nonspecific alkaline phosphatase diminishes craniosynostosis in one of two FGFR2C342Y\/+ mouse models of Crouzon syndrome. PLoS ONE, 15.","DOI":"10.1371\/journal.pone.0234073"},{"key":"ref_204","doi-asserted-by":"crossref","first-page":"1487","DOI":"10.1242\/dev.121.5.1487","article-title":"Tissue non-specific alkaline phosphatase is expressed in both embryonic and extraembryonic lineages during mouse embryogenesis but is not required for migration of primordial germ cells","volume":"121","author":"MacGregor","year":"1995","journal-title":"Development"},{"key":"ref_205","doi-asserted-by":"crossref","first-page":"116","DOI":"10.1002\/(SICI)1526-968X(200002)26:2<116::AID-GENE4>3.0.CO;2-X","article-title":"Targeted insertion of Cre recombinase into the TNAP gene: Excision in primordial germ cells","volume":"26","author":"Gertsenstein","year":"2000","journal-title":"Genesis"},{"key":"ref_206","doi-asserted-by":"crossref","unstructured":"Coticchio, G.A.D., and De Santis, L. (2013). Origin, Migration, and Proliferation of Human Primordial Germ Cells. Oogenesis, Springer.","DOI":"10.1007\/978-0-85729-826-3"},{"key":"ref_207","doi-asserted-by":"crossref","first-page":"863","DOI":"10.1016\/j.neuroscience.2007.07.064","article-title":"The ectonucleotidases alkaline phosphatase and nucleoside triphosphate diphosphohydrolase 2 are associated with subsets of progenitor cell populations in the mouse embryonic, postnatal and adult neurogenic zones","volume":"150","author":"Langer","year":"2007","journal-title":"Neuroscience"},{"key":"ref_208","unstructured":"Kaufman, M.H. (1992). The Atlas of Mouse Development, Academic Press."},{"key":"ref_209","doi-asserted-by":"crossref","first-page":"1034","DOI":"10.1242\/dev.085225","article-title":"Foxc1 controls the growth of the murine frontal bone rudiment by direct regulation of a Bmp response threshold of Msx2","volume":"140","author":"Sun","year":"2013","journal-title":"Development"},{"key":"ref_210","doi-asserted-by":"crossref","first-page":"116","DOI":"10.1038\/nrc2780","article-title":"Fibroblast growth factor signalling: From development to cancer","volume":"10","author":"Turner","year":"2010","journal-title":"Nat. Rev. Cancer"},{"key":"ref_211","doi-asserted-by":"crossref","first-page":"7158","DOI":"10.1038\/sj.onc.1210529","article-title":"Frequent activating FGFR2 mutations in endometrial carcinomas parallel germline mutations associated with craniosynostosis and skeletal dysplasia syndromes","volume":"26","author":"Pollock","year":"2007","journal-title":"Oncogene"},{"key":"ref_212","doi-asserted-by":"crossref","first-page":"831","DOI":"10.1111\/j.1432-0436.2007.00210.x","article-title":"Aberrant fibroblast growth factor receptor signaling in bladder and other cancers","volume":"75","author":"Chaffer","year":"2007","journal-title":"Differentiation"},{"key":"ref_213","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1016\/j.molonc.2012.02.004","article-title":"Discovery of small molecule cancer drugs: Successes, challenges and opportunities","volume":"6","author":"Hoelder","year":"2012","journal-title":"Mol. Oncol."},{"key":"ref_214","doi-asserted-by":"crossref","first-page":"1145","DOI":"10.1038\/ng2096","article-title":"RNA interference and inhibition of MEK-ERK signaling prevent abnormal skeletal phenotypes in a mouse model of craniosynostosis","volume":"39","author":"Shukla","year":"2007","journal-title":"Nat. Genet."},{"key":"ref_215","doi-asserted-by":"crossref","first-page":"18603","DOI":"10.1073\/pnas.0609157103","article-title":"Attenuation of signaling pathways stimulated by pathologically activated FGF-receptor 2 mutants prevents craniosynostosis","volume":"103","author":"Eswarakumar","year":"2006","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"ref_216","doi-asserted-by":"crossref","first-page":"215","DOI":"10.1227\/01.neu.0000243302.07308.d2","article-title":"A model for the pharmacological treatment of crouzon syndrome","volume":"59","author":"Perlyn","year":"2006","journal-title":"Neurosurgery"},{"key":"ref_217","doi-asserted-by":"crossref","first-page":"153","DOI":"10.1016\/j.tranon.2016.12.003","article-title":"Multi-Chemotherapeutic Schedules Containing the pan-FGFR Inhibitor ARQ 087 are Safe and Show Antitumor Activity in Different Xenograft Models","volume":"10","author":"Hall","year":"2017","journal-title":"Transl. Oncol."},{"key":"ref_218","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1038\/s41416-018-0334-0","article-title":"Derazantinib (ARQ 087) in advanced or inoperable FGFR2 gene fusion-positive intrahepatic cholangiocarcinoma","volume":"120","author":"Mazzaferro","year":"2019","journal-title":"Br. J. Cancer"},{"key":"ref_219","doi-asserted-by":"crossref","first-page":"57","DOI":"10.1016\/j.bone.2017.08.016","article-title":"ARQ 087 inhibits FGFR signaling and rescues aberrant cell proliferation and differentiation in experimental models of craniosynostoses and chondrodysplasias caused by activating mutations in FGFR1, FGFR2 and FGFR3","volume":"105","author":"Balek","year":"2017","journal-title":"Bone"},{"key":"ref_220","doi-asserted-by":"crossref","first-page":"4585","DOI":"10.1021\/jm100054f","article-title":"Heat Shock Protein 70 (Hsp70) as an Emerging Drug Target","volume":"53","author":"Evans","year":"2010","journal-title":"J. Med. Chem."},{"key":"ref_221","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1002\/hep.30127","article-title":"HSP90 Inhibition Drives Degradation of FGFR2 Fusion Proteins: Implications for Treatment of Cholangiocarcinoma","volume":"69","author":"Lamberti","year":"2019","journal-title":"Hepatology"},{"key":"ref_222","doi-asserted-by":"crossref","first-page":"152","DOI":"10.1016\/j.bbrc.2016.07.111","article-title":"The E3 ubiquitin ligase CHIP selectively regulates mutant epidermal growth factor receptor by ubiquitination and degradation","volume":"479","author":"Chung","year":"2016","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"ref_223","doi-asserted-by":"crossref","first-page":"17193","DOI":"10.3390\/ijms160817193","article-title":"Protein Folding and Mechanisms of Proteostasis","volume":"16","year":"2015","journal-title":"Int. J. Mol. Sci."},{"key":"ref_224","doi-asserted-by":"crossref","first-page":"537","DOI":"10.1038\/nrc2887","article-title":"Targeting the dynamic HSP90 complex in cancer","volume":"10","author":"Trepel","year":"2010","journal-title":"Nat. Rev. Cancer"},{"key":"ref_225","doi-asserted-by":"crossref","first-page":"36259","DOI":"10.1074\/jbc.M402469200","article-title":"Cbl-mediated Degradation of Lyn and Fyn Induced by Constitutive Fibroblast Growth Factor Receptor-2 Activation Supports Osteoblast Differentiation","volume":"279","author":"Kaabeche","year":"2004","journal-title":"J. Biol. Chem."},{"key":"ref_226","doi-asserted-by":"crossref","first-page":"1223","DOI":"10.1242\/jcs.01679","article-title":"Cbl-mediated ubiquitination of 5 integrin subunit mediates fibronectin-dependent osteoblast detachment and apoptosis induced by FGFR2 activation","volume":"118","author":"Kaabeche","year":"2005","journal-title":"J. Cell Sci."},{"key":"ref_227","doi-asserted-by":"crossref","first-page":"27292","DOI":"10.1074\/jbc.M600448200","article-title":"Intracellular Retention, Degradation, and Signaling of Glycosylation-deficient FGFR2 and Craniosynostosis Syndrome-associated FGFR2C278F","volume":"281","author":"Hatch","year":"2006","journal-title":"J. Biol. Chem."},{"key":"ref_228","doi-asserted-by":"crossref","first-page":"32751","DOI":"10.1074\/jbc.M803183200","article-title":"The Boston-type Craniosynostosis Mutation MSX2 (P148H) Results in Enhanced Susceptibility of MSX2 to Ubiquitin-dependent Degradation","volume":"283","author":"Yoon","year":"2008","journal-title":"J. Biol. Chem."},{"key":"ref_229","doi-asserted-by":"crossref","first-page":"eaaw0946","DOI":"10.1126\/sciadv.aaw0946","article-title":"p63 establishes epithelial enhancers at critical craniofacial development genes","volume":"5","author":"Lan","year":"2019","journal-title":"Sci. Adv."},{"key":"ref_230","doi-asserted-by":"crossref","first-page":"eaas9536","DOI":"10.1126\/science.aas9536","article-title":"Spatiotemporal structure of cell fate decisions in murine neural crest","volume":"364","author":"Soldatov","year":"2019","journal-title":"Science"},{"key":"ref_231","doi-asserted-by":"crossref","first-page":"260","DOI":"10.1136\/jmedgenet-2016-104215","article-title":"Diagnostic value of exome and whole genome sequencing in craniosynostosis","volume":"54","author":"Miller","year":"2016","journal-title":"J. Med. Genet."},{"key":"ref_232","first-page":"e60051","article-title":"Dissection, Culture and Analysis of Primary Cranial Neural Crest Cells from Mouse for the Study of Neural Crest Cell Delamination and Migration","volume":"2019","author":"Malagon","year":"2019","journal-title":"J. Vis. Exp."},{"key":"ref_233","doi-asserted-by":"crossref","first-page":"3069","DOI":"10.1089\/scd.2012.0155","article-title":"A Stable Cranial Neural Crest Cell Line from Mouse","volume":"21","author":"Ishii","year":"2012","journal-title":"Stem Cells Dev."}],"container-title":["Journal of Developmental Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2221-3759\/8\/3\/18\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T10:08:19Z","timestamp":1760177299000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2221-3759\/8\/3\/18"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,9,9]]},"references-count":233,"journal-issue":{"issue":"3","published-online":{"date-parts":[[2020,9]]}},"alternative-id":["jdb8030018"],"URL":"https:\/\/doi.org\/10.3390\/jdb8030018","relation":{},"ISSN":["2221-3759"],"issn-type":[{"value":"2221-3759","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,9,9]]}}}