...Kuo-Chang Tseng; J. Gage Crump ABSTRACT The evolution of a unique craniofacial complex in vertebrates made possible new ways of breathing, eating, communicating and sensing the environment. The head and face develop through interactions of all three germ layers, the endoderm, ectoderm and mesoderm...

... expressed genes within the ciliome that display tissue and temporal specificity. Primary cilia Tissue heterogeneity Craniofacial Skeletogenesis Ciliopathy National Institute of General Medical Sciences http://dx.doi.org/10.13039/100000057 R01 GM124251 National Institutes...

... a highly penetrant cleft palate phenotype, shortened limbs compared with wild type and perinatal lethality. Fzd2 D4 craniofacial tissues indicated decreased canonical Wnt signaling. In utero treatment with IIIC3a (a DKK inhibitor) normalized the limb lengths in Fzd2 D4 homozygotes. The in vivo replication...

... epithelial migration driven by pulsatile actomyosin contractility. Palate Collective cell migration Apoptosis Cell extrusion Tgfβ Cleft palate Cleft lip Craniofacial Live imaging Fusion Actomyosin Cell adhesion Non-muscle myosin II NMIIA Mouse National Institute of Dental...

... bone formation, alterations in which contribute to congenital birth defects. Here, we demonstrate that transcription factor and histone methyltransferase proteins Prdm3 and Prdm16 control the differentiation switch of cranial NCCs to craniofacial cartilage. Loss of either paralog results in hypoplastic...

... of order/disorder as well as symmetry/asymmetry, revealing developmental instabilities that are part of normal morphogenesis. Craniofacial Avian embryo Frontonasal Fluctuating asymmetry Mathematical analysis Live cell imaging Organ culture Actomyosin Facial morphogenesis requires...

... comparison (morphometrics) traditionally uses manually located landmarks and is limited by landmark number and operator accuracy. Here, we apply a landmark-free method to characterise the craniofacial skeletal phenotype of the Dp1Tyb mouse model of Down syndrome and a population of the Diversity Outbred (DO...

... complementary models indicate that disruption of Irf6 and Esrp1/2 in mouse and zebrafish results in orofacial clefts, with an aberrant mesenchymal/epithelial cell population identified in the cleft of the esrp1/2 zebrafish mutant. IRF6 ESRP1 Craniofacial Cleft Development Development...

...Karl B. Shpargel; Cassidy L. Mangini; Guojia Xie; Kai Ge; Terry Magnuson; Sally Dunwoodie; John Wallingford ABSTRACT Kabuki syndrome (KS) is a congenital craniofacial disorder resulting from mutations in the KMT2D histone methylase (KS1) or the UTX histone demethylase (KS2). With small cohorts...

...Ceilidh Marchant; Peter Anderson; Quenten Schwarz; Sophie Wiszniak ABSTRACT Craniofacial development is a complex morphogenic process that requires highly orchestrated interactions between multiple cell types. Blood vessel-derived angiocrine factors are known to promote proliferation...

...-metamorphic Xenopus laevis tadpoles to self-correct malformed craniofacial tissues. We found that tadpoles can adaptively improve and normalize abnormal craniofacial morphology caused by numerous developmental perturbations. We then investigated the tissue-level and molecular mechanisms that mediate the self...

... differentiation. During normal zebrafish craniofacial development, barx1 and then sox9a mark CNCs in pre-cartilage condensations ( Nichols et al., 2013 ; Barske et al., 2016 ). Sox9a then induces a number of cartilage differentiation genes, including those encoding for the extracellular matrix components...

... for craniofacial development, how genetic programs drive this regionalization remains incompletely understood. Here we use combinatorial labeling of zebrafish cranial neural crest-derived cells (CNCCs) to define global gene expression along the dorsoventral axis of the developing arches. Intersection of region...

.... Endothelin Craniofacial Neural crest cell Hinge and caps Knockout mouse Development of the vertebrate face requires the coordinated regulation of patterning cues throughout the pharyngeal arches of the developing embryo. Populated by cranial neural crest cells (NCCs) originating in the dorsal...

... read and commented on it Funding This work was supported by a Cincinnati Children's Hospital Medical Center Trustee Award (to Y.L.); and by grants from the National Institutes of Health National Institute of Dental and Craniofacial Research [R03DE023864 to Y.L., R01DE013681 to R.J.]. Deposited...

... during pharyngeal pouch formation in the zebrafish head. Eph-ephrin PAK Branching morphogenesis Craniofacial Pharyngeal pouches Zebrafish During development, epithelial tissues undergo morphogenesis to form elaborately branched organs. Whereas epithelial cells are characterized...

.... Although phenotypically similar to talpid 3 , talpid 2 has a distinct facial phenotype and an unknown cellular, molecular and genetic basis. We set out to determine the etiology of the craniofacial phenotype of this mutant. We confirmed that primary cilia were disrupted in talpid 2 mutants. Molecularly, we...

... craniofacial tendon and ligament progenitors are neural crest derived, as in mammals. Cranial and fin tendon progenitors can be induced in the absence of differentiated muscle or cartilage, although neighboring muscle and cartilage are required for tendon cell maintenance and organization, respectively...

...-type and mutant craniofacial structures, with live images (F-I), dissected neurocranium (J-M), flat-mounted lower jaw structures ( R-U ) at 10× magnification and ethmoid plate (N-Q) at 40× magnification. The R84C cleft phenotype is the most severe (arrowheads, O). R84H has a milder cleft phenotype...

... might also account for the high phenotypic variation observed in human FRAS1 patients. fras1 Zebrafish Craniofacial Fraser syndrome Developmental instability Human FRAS1 lesions cause Fraser syndrome ( McGregor et al., 2003 ; Slavotinek et al., 2006 ; van Haelst et al., 2008...