Gene Symbol: Foxc1
Description: forkhead box C1
Alias: FREAC3, Fkh1, Mf1, Mf4, fkh-1, frkhda, forkhead box protein C1, FREAC-3, MF-1, congenital hydrocephalus, forkhead-related protein FKHL7, forkhead-related transcription factor 3, mesoderm/mesenchyme forkhead 1, transcription factor FKH-1
Species: mouse
Products:     Foxc1

Top Publications

  1. Seo S, Kume T. Forkhead transcription factors, Foxc1 and Foxc2, are required for the morphogenesis of the cardiac outflow tract. Dev Biol. 2006;296:421-36 pubmed
    Previous studies have shown that Foxc1 and Foxc2, closely related Fox transcription factors, have interactive roles in cardiovascular development. However, little is known about their functional overlap during early heart morphogenesis...
  2. Rice R, Rice D, Olsen B, Thesleff I. Progression of calvarial bone development requires Foxc1 regulation of Msx2 and Alx4. Dev Biol. 2003;262:75-87 pubmed
    ..b>Congenital hydrocephalus (ch) mutant mice lack the forkhead/winged helix transcription factor Foxc1...
  3. Sommer P, Napier H, Hogan B, Kidson S. Identification of Tgf beta1i4 as a downstream target of Foxc1. Dev Growth Differ. 2006;48:297-308 pubmed
    Craniofacial development is severely affected by null mutations in Foxc1, indicating a multifunctional role for Foxc1 in ocular, maxilla and mandible, skull and facial gland development...
  4. Inoue T, Ogawa M, Mikoshiba K, Aruga J. Zic deficiency in the cortical marginal zone and meninges results in cortical lamination defects resembling those in type II lissencephaly. J Neurosci. 2008;28:4712-25 pubmed publisher
    ..These findings indicate that the Zic genes play critical roles in cortical development through regulating the proliferation of meningeal cells and the pial BM assembly. ..
  5. Mears A, Jordan T, Mirzayans F, Dubois S, Kume T, Parlee M, et al. Mutations of the forkhead/winged-helix gene, FKHL7, in patients with Axenfeld-Rieger anomaly. Am J Hum Genet. 1998;63:1316-28 pubmed
    ..Abnormalities of this locus lead to glaucoma. FKHL7 (also called "FREAC3"), a member of the forkhead/winged-helix transcription-factor family, has also been mapped to 6p25...
  6. Seo S, Fujita H, Nakano A, Kang M, Duarte A, Kume T. The forkhead transcription factors, Foxc1 and Foxc2, are required for arterial specification and lymphatic sprouting during vascular development. Dev Biol. 2006;294:458-70 pubmed
    ..Here, we show that mouse embryos compound mutant for Foxc1 and Foxc2, two closely related Fox transcription factors, exhibit arteriovenous malformations and lack of ..
  7. Berry F, Saleem R, Walter M. FOXC1 transcriptional regulation is mediated by N- and C-terminal activation domains and contains a phosphorylated transcriptional inhibitory domain. J Biol Chem. 2002;277:10292-7 pubmed
    Mutations in the FOXC1 gene result in Axenfeld-Rieger malformations of the anterior segment of the eye and lead to an increased susceptibility of glaucoma...
  8. Sasman A, Nassano Miller C, Shim K, Koo H, Liu T, Schultz K, et al. Generation of conditional alleles for Foxc1 and Foxc2 in mice. Genesis. 2012;50:766-74 pubmed publisher
    The Forkhead box transcription factors, Foxc1 and Foxc2, are crucial for development of the eye, cardiovascular network, and other physiological systems, but their cell-type specific and postdevelopmental functions are unknown, in part ..
  9. Saleem R, Banerjee Basu S, Berry F, Baxevanis A, Walter M. Analyses of the effects that disease-causing missense mutations have on the structure and function of the winged-helix protein FOXC1. Am J Hum Genet. 2001;68:627-41 pubmed
    Five missense mutations of the winged-helix FOXC1 transcription factor, found in patients with Axenfeld-Rieger (AR) malformations, were investigated for their effects on FOXC1 structure and function...

More Information


  1. Zarbalis K, Siegenthaler J, Choe Y, May S, Peterson A, Pleasure S. Cortical dysplasia and skull defects in mice with a Foxc1 allele reveal the role of meningeal differentiation in regulating cortical development. Proc Natl Acad Sci U S A. 2007;104:14002-7 pubmed
    We report the identification of a hypomorphic mouse allele for Foxc1 (Foxc1(hith)) that survives into adulthood revealing previously unknown roles for Foxc1 in development of the skull and cerebral cortex...
  2. Smith R, Zabaleta A, Kume T, Savinova O, Kidson S, Martin J, et al. Haploinsufficiency of the transcription factors FOXC1 and FOXC2 results in aberrant ocular development. Hum Mol Genet. 2000;9:1021-32 pubmed
    ..The forkhead transcription factor genes Foxc1 (formerly Mf1 ) and Foxc2 (formerly Mfh1 ) are expressed in the mesenchyme from which the ocular drainage ..
  3. Kume T, Deng K, Hogan B. Murine forkhead/winged helix genes Foxc1 (Mf1) and Foxc2 (Mfh1) are required for the early organogenesis of the kidney and urinary tract. Development. 2000;127:1387-95 pubmed
    ..Foxc1/Mf1 is disrupted in the mutant, congenital hydrocephalus (Foxc1/Mf1(ch)), which has multiple developmental defects...
  4. Winnier G, Kume T, Deng K, Rogers R, Bundy J, Raines C, et al. Roles for the winged helix transcription factors MF1 and MFH1 in cardiovascular development revealed by nonallelic noncomplementation of null alleles. Dev Biol. 1999;213:418-31 pubmed
    The murine Mf1 and Mfh1 genes have overlapping patterns of expression in the embryo and encode forkhead/winged helix transcription factors with virtually identical DNA binding domains...
  5. Hong H, Lass J, Chakravarti A. Pleiotropic skeletal and ocular phenotypes of the mouse mutation congenital hydrocephalus (ch/Mf1) arise from a winged helix/forkhead transcriptionfactor gene. Hum Mol Genet. 1999;8:625-37 pubmed
    b>Congenital hydrocephalus is an etiologically diverse, poorly understood, but relatively common birth defect. Most human cases are sporadic with familial forms showing considerable phenotypic and etiologic heterogeneity...
  6. Rice R, Rice D, Thesleff I. Foxc1 integrates Fgf and Bmp signalling independently of twist or noggin during calvarial bone development. Dev Dyn. 2005;233:847-52 pubmed
    ..We have shown previously that the winged helix transcription factor Foxc1, which is necessary for calvarial bone development, is required for the Bmp regulation of Msx2...
  7. Kume T, Jiang H, Topczewska J, Hogan B. The murine winged helix transcription factors, Foxc1 and Foxc2, are both required for cardiovascular development and somitogenesis. Genes Dev. 2001;15:2470-82 pubmed
    The murine Foxc1/Mf1 and Foxc2/Mfh1 genes encode closely related forkhead/winged helix transcription factors with overlapping expression in the forming somites and head mesoderm and endothelial and mesenchymal cells of the developing ..
  8. Zarbalis K, May S, Shen Y, Ekker M, Rubenstein J, Peterson A. A focused and efficient genetic screening strategy in the mouse: identification of mutations that disrupt cortical development. PLoS Biol. 2004;2:E219 pubmed
    ..Our results also highlight the utility of focused screens in the mouse, in addition to the large-scale and broadly targeted screens that are being carried out at mutagenesis centers. ..
  9. Wilm B, James R, Schultheiss T, Hogan B. The forkhead genes, Foxc1 and Foxc2, regulate paraxial versus intermediate mesoderm cell fate. Dev Biol. 2004;271:176-89 pubmed
    ..Here, we provide evidence that the forkhead genes, Foxc1 and Foxc2, play a role in the specification of mesoderm to paraxial versus intermediate fates...
  10. Yamagishi H, Maeda J, Hu T, McAnally J, Conway S, Kume T, et al. Tbx1 is regulated by tissue-specific forkhead proteins through a common Sonic hedgehog-responsive enhancer. Genes Dev. 2003;17:269-81 pubmed
    ..Foxa2, Foxc1, or Foxc2 could bind and activate transcription through the critical cis-element upstream of Tbx1, and Foxc ..
  11. Kume T, Deng K, Winfrey V, Gould D, Walter M, Hogan B. The forkhead/winged helix gene Mf1 is disrupted in the pleiotropic mouse mutation congenital hydrocephalus. Cell. 1998;93:985-96 pubmed
    ..eye defects, and multiple skeletal abnormalities identical to those of the classical mutant, congenital hydrocephalus. We show that congenital hydrocephalus involves a point mutation in Mf1, generating a truncated protein ..
  12. Vivatbutsiri P, Ichinose S, Hytönen M, Sainio K, Eto K, Iseki S. Impaired meningeal development in association with apical expansion of calvarial bone osteogenesis in the Foxc1 mutant. J Anat. 2008;212:603-11 pubmed publisher
    Loss of function of the mouse forkhead/winged helix transcription factor Foxc1 induces congenital hydrocephalus and impaired skull bone development due to failure of apical expansion of the bone...
  13. Pierrou S, Hellqvist M, Samuelsson L, Enerback S, Carlsson P. Cloning and characterization of seven human forkhead proteins: binding site specificity and DNA bending. EMBO J. 1994;13:5002-12 pubmed
    ..Applying a circular permutation assay, we show that binding of FREAC proteins to their cognate sites results in bending of the DNA at an angle of 80-90 degrees. ..
  14. Siegenthaler J, Ashique A, Zarbalis K, Patterson K, Hecht J, KANE M, et al. Retinoic acid from the meninges regulates cortical neuron generation. Cell. 2009;139:597-609 pubmed publisher
    ..We took advantage of Foxc1 mutant mice with defects in forebrain meningeal formation...
  15. Kidson S, Kume T, Deng K, Winfrey V, Hogan B. The forkhead/winged-helix gene, Mf1, is necessary for the normal development of the cornea and formation of the anterior chamber in the mouse eye. Dev Biol. 1999;211:306-22 pubmed
    b>Mf1, which encodes a winged-helix/forkhead transcription factor, is the murine homolog of human FKHL7, mutated in individuals with autosomal dominant inherited dysgenesis of the anterior segment of the eye (Axenfeld-Reiger anomaly)...
  16. Hayashi H, Kume T. Forkhead transcription factors regulate expression of the chemokine receptor CXCR4 in endothelial cells and CXCL12-induced cell migration. Biochem Biophys Res Commun. 2008;367:584-9 pubmed publisher
    b>Foxc1 and Foxc2 transcription factors are required for vascular development...
  17. Choe Y, Siegenthaler J, Pleasure S. A cascade of morphogenic signaling initiated by the meninges controls corpus callosum formation. Neuron. 2012;73:698-712 pubmed publisher
    ..The effects of GDF5 are in turn under the control of a soluble GDF5 inhibitor, Dan, made by the meninges. Thus, the meninges and medial neocortex use a cascade of signals to regulate corpus callosum development. ..
  18. Aldinger K, Lehmann O, Hudgins L, Chizhikov V, Bassuk A, Ades L, et al. FOXC1 is required for normal cerebellar development and is a major contributor to chromosome 6p25.3 Dandy-Walker malformation. Nat Genet. 2009;41:1037-42 pubmed publisher
    ..Here we characterize a second DWM-linked locus on 6p25.3, showing that deletions or duplications encompassing FOXC1 are associated with cerebellar and posterior fossa malformations including cerebellar vermis hypoplasia (CVH), ..
  19. Breen M, Richardson R, Bondareff W, Weinstein H. Acidic glycosaminoglycans in developing sterno-costal cartilage of the hydrocephalic (ch+-ch+) mouse. Biochim Biophys Acta. 1973;304:828-36 pubmed
  20. Lagha M, Brunelli S, Messina G, Cumano A, Kume T, Relaix F, et al. Pax3:Foxc2 reciprocal repression in the somite modulates muscular versus vascular cell fate choice in multipotent progenitors. Dev Cell. 2009;17:892-9 pubmed publisher
    ..This provides insight into how cell fate choices are orchestrated by these lineage genes in the dermomyotome. ..
  21. Gould D, John S. Anterior segment dysgenesis and the developmental glaucomas are complex traits. Hum Mol Genet. 2002;11:1185-93 pubmed
    ..To minimize overlap with other reviews in this issue and elsewhere, we highlight the complex, multifactorial nature of these diseases and recent advances using mice. ..
  22. Gitler A, Lu M, Epstein J. PlexinD1 and semaphorin signaling are required in endothelial cells for cardiovascular development. Dev Cell. 2004;7:107-16 pubmed
    ..Thus, molecular programs that mediate axon guidance in the central nervous system also function in endothelial cells to orchestrate critical aspects of cardiac morphogenesis. ..
  23. Motojima M, Tanimoto S, Ohtsuka M, Matsusaka T, Kume T, Abe K. Characterization of Kidney and Skeleton Phenotypes of Mice Double Heterozygous for Foxc1 and Foxc2. Cells Tissues Organs. 2016;201:380-9 pubmed publisher
    b>Foxc1 and Foxc2 play key roles in mouse development. Foxc1 mutant mice develop duplex kidneys with double ureters, and lack calvarial and sternal bones...
  24. Zacharias A, Gage P. Canonical Wnt/?-catenin signaling is required for maintenance but not activation of Pitx2 expression in neural crest during eye development. Dev Dyn. 2010;239:3215-25 pubmed publisher
    ..Other key transcription factors in the neural crest, including Foxc1, do not require intact canonical Wnt signaling.
  25. Wang J, Xiao Y, Hsu C, Martinez Traverso I, Zhang M, Bai Y, et al. Yap and Taz play a crucial role in neural crest-derived craniofacial development. Development. 2016;143:504-15 pubmed publisher
    ..RNA-sequencing data indicated that Yap and Taz regulate genes encoding Fox transcription factors, specifically Foxc1. Proliferation was reduced in the branchial arch mesenchyme of Yap and Taz CNC conditional knockout (CKO) embryos...
  26. Hopkins A, Mirzayans F, Berry F. Foxc1 Expression in Early Osteogenic Differentiation Is Regulated by BMP4-SMAD Activity. J Cell Biochem. 2016;117:1707-17 pubmed publisher
    b>FOXC1 is an important regulator of the initial steps in intramembranous and endochondral ossification processes...
  27. Mayeuf Louchart A, Montarras D, Bodin C, Kume T, Vincent S, Buckingham M. Endothelial cell specification in the somite is compromised in Pax3-positive progenitors of Foxc1/2 conditional mutants, with loss of forelimb myogenesis. Development. 2016;143:872-9 pubmed publisher
    ..b>Foxc1 has overlapping functions with Foxc2...
  28. Hayashi H, Sano H, Seo S, Kume T. The Foxc2 transcription factor regulates angiogenesis via induction of integrin beta3 expression. J Biol Chem. 2008;283:23791-800 pubmed publisher
    ..Taken together, these results suggest that Foxc2 directly induces Itgb3 expression and regulates angiogenesis by Itgb3-mediated endothelial cell adhesion and migration. ..
  29. Matt N, Dupé V, Garnier J, Dennefeld C, Chambon P, Mark M, et al. Retinoic acid-dependent eye morphogenesis is orchestrated by neural crest cells. Development. 2005;132:4789-800 pubmed control: (1) the remodeling of the POM through activation of Eya2-related apoptosis; (2) the expression of Foxc1 and Pitx2, which play crucial roles in anterior eye segment development; and (3) the growth of the ventral retina...
  30. Dagenais S, Hartsough R, Erickson R, Witte M, Butler M, Glover T. Foxc2 is expressed in developing lymphatic vessels and other tissues associated with lymphedema-distichiasis syndrome. Gene Expr Patterns. 2004;4:611-9 pubmed
    ..mouse development using immunohistochemistry, and examined its expression in lymphatics compared to its paralog Foxc1 and to Vegfr-3, Prox1 and other lymphatic and blood vascular proteins...
  31. Gitler A, Lu M, Jiang Y, Epstein J, Gruber P. Molecular markers of cardiac endocardial cushion development. Dev Dyn. 2003;228:643-50 pubmed
  32. Hiemisch H, Monaghan A, Schutz G, Kaestner K. Expression of the mouse Fkh1/Mf1 and Mfh1 genes in late gestation embryos is restricted to mesoderm derivatives. Mech Dev. 1998;73:129-32 pubmed
    We have compared the expression patterns of the mouse Forkhead homologue 1/ mesoderm/mesenchyme forkhead 1 (Fkh1/Mf1) gene with that of the highly related winged helix gene Mfh1 in late gestation mouse embryos...
  33. Fatima A, Wang Y, Uchida Y, Norden P, Liu T, Culver A, et al. Foxc1 and Foxc2 deletion causes abnormal lymphangiogenesis and correlates with ERK hyperactivation. J Clin Invest. 2016;126:2437-51 pubmed publisher
    ..dominantly associated with late-onset lymphedema; however, the precise role of FOXC2 and a closely related factor, FOXC1, in the lymphatic system remains largely unknown...
  34. Winnier G, Hargett L, Hogan B. The winged helix transcription factor MFH1 is required for proliferation and patterning of paraxial mesoderm in the mouse embryo. Genes Dev. 1997;11:926-40 pubmed
    ..g., mtwist, mf1, scleraxis, and alpha1(II) collagen] is seen in the anterior of homozygous mutants...
  35. Kochilas L, Merscher Gomez S, Lu M, Potluri V, Liao J, Kucherlapati R, et al. The role of neural crest during cardiac development in a mouse model of DiGeorge syndrome. Dev Biol. 2002;251:157-66 pubmed
    ..Based on our studies, we propose that Lgdel genes are required for the expression of soluble signals that regulate neural crest cell differentiation. ..
  36. Mattiske D, Sommer P, Kidson S, Hogan B. The role of the forkhead transcription factor, Foxc1, in the development of the mouse lacrimal gland. Dev Dyn. 2006;235:1074-80 pubmed
    The lacrimal gland produces secretions that lubricate and protect the cornea of the eye. Foxc1 encodes a forkhead/winged helix transcription factor required for the development of many embryonic organs...
  37. Siegenthaler J, Choe Y, Patterson K, Hsieh I, Li D, Jaminet S, et al. Foxc1 is required by pericytes during fetal brain angiogenesis. Biol Open. 2013;2:647-59 pubmed publisher
    ..Here we show that the forkhead transcription factor Foxc1 is expressed by brain pericytes during development and is critical for pericyte regulation of vascular development ..
  38. Libby R, Smith R, Savinova O, Zabaleta A, Martin J, Gonzalez F, et al. Modification of ocular defects in mouse developmental glaucoma models by tyrosinase. Science. 2003;299:1578-81 pubmed
    ..Tyr also modified the drainage structure dysgenesis in mice with a mutant Foxc1 gene, which is also involved in PCG...
  39. Haldipur P, Dang D, Aldinger K, Janson O, Guimiot F, Adle Biasette H, et al. Phenotypic outcomes in Mouse and Human Foxc1 dependent Dandy-Walker cerebellar malformation suggest shared mechanisms. elife. 2017;6: pubmed publisher
    i>FOXC1 loss contributes to Dandy-Walker malformation (DWM), a common human cerebellar malformation...
  40. Mitchell E, Taylor D, Woods K, Davis M, Nelson A, Teasdale R, et al. Differential gene expression in the developing mouse ureter. Gene Expr Patterns. 2006;6:519-38 pubmed
    ..surrounded by ureteric mesenchyme, genes previously reported to be expressed in the ureteric mesenchyme, foxC1 and foxC2 were upregulated. By E15...
  41. Clevidence D, Overdier D, Tao W, Qian X, Pani L, Lai E, et al. Identification of nine tissue-specific transcription factors of the hepatocyte nuclear factor 3/forkhead DNA-binding-domain family. Proc Natl Acad Sci U S A. 1993;90:3948-52 pubmed
    ..The HNF-3/fkh motif therefore comprises a large gene family of transcription factors that play a role in tissue-specific gene regulation and development. ..
  42. Murashima A, Akita H, Okazawa M, Kishigami S, Nakagata N, Nishinakamura R, et al. Midline-derived Shh regulates mesonephric tubule formation through the paraxial mesoderm. Dev Biol. 2014;386:216-26 pubmed publisher
    ..These data demonstrate the essential role of midline-derived Shh in local tissue morphogenesis and differentiation. ..
  43. Qu X, Pan Y, Carbe C, Powers A, Grobe K, Zhang X. Glycosaminoglycan-dependent restriction of FGF diffusion is necessary for lacrimal gland development. Development. 2012;139:2730-9 pubmed publisher
    ..Taken together, these results demonstrate that mesenchymal GAG controls lacrimal gland induction by restricting the diffusion of Fgf10...
  44. Sizemore G, Sizemore S, Pal B, Booth C, Seachrist D, Abdul Karim F, et al. FOXC1 is enriched in the mammary luminal progenitor population, but is not necessary for mouse mammary ductal morphogenesis. Biol Reprod. 2013;89:10 pubmed publisher
    Expression of FOXC1, a forkhead box transcription factor, correlates with the human basal-like breast cancer (BLBC) subtype, and functional analyses have revealed its importance for in vitro invasiveness of BLBC cells...
  45. Seo S, Chen L, Liu W, Zhao D, Schultz K, Sasman A, et al. Foxc1 and Foxc2 in the Neural Crest Are Required for Ocular Anterior Segment Development. Invest Ophthalmol Vis Sci. 2017;58:1368-1377 pubmed publisher
    ..Similarly, we also generated compound NC-specific mutations of Foxc2 and a closely related gene, Foxc1 (NC-Foxc1-/-;NC-Foxc2-/-) in mice...
  46. Chen L, Martino V, Dombkowski A, Williams T, WEST MAYS J, Gage P. AP-2β Is a Downstream Effector of PITX2 Required to Specify Endothelium and Establish Angiogenic Privilege During Corneal Development. Invest Ophthalmol Vis Sci. 2016;57:1072-81 pubmed publisher
    ..Therefore, additional PITX2-dependent factors likely regulate these latter processes during embryonic development. These results extend our understanding of the genetic mechanisms regulating cornea development. ..
  47. Garg A, Bansal M, Gotoh N, Feng G, Zhong J, Wang F, et al. Alx4 relays sequential FGF signaling to induce lacrimal gland morphogenesis. PLoS Genet. 2017;13:e1007047 pubmed publisher
    ..Inactivation of ALX4/Alx4 causes lacrimal gland aplasia in both human and mouse. These results reveal a key role of Alx4 in mediating FGF-Shp2-FGF signaling in the neural crest for lacrimal gland development. ..
  48. Yoshida M, Hata K, Takashima R, Ono K, Nakamura E, Takahata Y, et al. The transcription factor Foxc1 is necessary for Ihh-Gli2-regulated endochondral ossification. Nat Commun. 2015;6:6653 pubmed publisher
    ..Here by using in vivo microarray analysis, we identify forkhead box C1 (Foxc1) as a transcriptional partner of Gli2...
  49. Furumoto T, Miura N, Akasaka T, Mizutani Koseki Y, Sudo H, Fukuda K, et al. Notochord-dependent expression of MFH1 and PAX1 cooperates to maintain the proliferation of sclerotome cells during the vertebral column development. Dev Biol. 1999;210:15-29 pubmed
    ..Thus, both the Mfh1 and the Pax1 gene products cooperate to mediate Sonic hedgehog-dependent proliferation of sclerotome cells. ..
  50. Huang J, Dattilo L, Rajagopal R, Liu Y, Kaartinen V, Mishina Y, et al. FGF-regulated BMP signaling is required for eyelid closure and to specify conjunctival epithelial cell fate. Development. 2009;136:1741-50 pubmed publisher
    ..Transcripts encoding Foxc1 and Foxc2, which were previously shown to be necessary for eyelid closure, were not detectable in Smad4(CKO) ..
  51. Miura N, Wanaka A, Tohyama M, Tanaka K. MFH-1, a new member of the fork head domain family, is expressed in developing mesenchyme. FEBS Lett. 1993;326:171-6 pubmed
    ..Our results suggest that MFH-1 might be involved in the formation of special mesenchymal tissues. ..
  52. Wang B, Weidenfeld J, Lu M, Maika S, Kuziel W, Morrisey E, et al. Foxp1 regulates cardiac outflow tract, endocardial cushion morphogenesis and myocyte proliferation and maturation. Development. 2004;131:4477-87 pubmed
  53. Hiemisch H, Schutz G, Kaestner K. The mouse Fkh1/Mf1 gene: cDNA sequence, chromosomal localization and expression in adult tissues. Gene. 1998;220:77-82 pubmed
    ..describe the chromosomal position, start site of transcription, sequence and adult expression pattern of the mouse Fkh1/Mf1 (Forkhead homologue 1/mesoderm/mesenchyme forkhead 1) gene...
  54. Teo A, Arnold S, Trotter M, Brown S, Ang L, Chng Z, et al. Pluripotency factors regulate definitive endoderm specification through eomesodermin. Genes Dev. 2011;25:238-50 pubmed publisher
    ..Together, these results provide for the first time a comprehensive molecular model connecting the transition from pluripotency to endoderm specification during mammalian development. ..
  55. Nishita M, Qiao S, Miyamoto M, Okinaka Y, Yamada M, Hashimoto R, et al. Role of Wnt5a-Ror2 signaling in morphogenesis of the metanephric mesenchyme during ureteric budding. Mol Cell Biol. 2014;34:3096-105 pubmed publisher
    ..These results indicate an important role of Wnt5a-Ror2 signaling in morphogenesis of the MM to ensure proper epithelial tubular formation of the UB required for kidney development. ..
  56. Labosky P, Winnier G, Sasaki H, Blessing M, Hogan B. The chromosomal mapping of four genes encoding winged helix proteins expressed early in mouse development. Genomics. 1996;34:241-5 pubmed
    ..b>Mf1 was assigned to mouse Chromosome 8, Mf2 to Chromosome 4, Mf3 to Chromosome 9, and Mf4 to Chromosome 13...
  57. Hoshino T, Shimizu R, Ohmori S, Nagano M, Pan X, Ohneda O, et al. Reduced BMP4 abundance in Gata2 hypomorphic mutant mice result in uropathies resembling human CAKUT. Genes Cells. 2008;13:159-70 pubmed publisher
    ..These experiments taken together implicate GATA-2 as a direct regulator of Bmp4 transcription. The pathophysiology described in Gata2 hypomorphic mutant animals resembles human congenital anomalies of the kidney and urinary tract. ..
  58. Gage P, Qian M, Wu D, Rosenberg K. The canonical Wnt signaling antagonist DKK2 is an essential effector of PITX2 function during normal eye development. Dev Biol. 2008;317:310-24 pubmed publisher
    ..We further propose a model placing PITX2 as an essential integration node between retinoic acid and canonical Wnt signaling during eye development. ..
  59. Haldipur P, Gillies G, Janson O, Chizhikov V, Mithal D, Miller R, et al. Foxc1 dependent mesenchymal signalling drives embryonic cerebellar growth. elife. 2014;3: pubmed publisher
    Loss of Foxc1 is associated with Dandy-Walker malformation, the most common human cerebellar malformation characterized by cerebellar hypoplasia and an enlarged posterior fossa and fourth ventricle...
  60. Sasaki H, Hogan B. Differential expression of multiple fork head related genes during gastrulation and axial pattern formation in the mouse embryo. Development. 1993;118:47-59 pubmed
    ..A model is proposed linking FD gene expression with gastrulation events in the mouse. ..
  61. Zhang Z, Leclerc D, Gravel R, Rozen R. The methionine synthase (Mtr) gene maps to proximal mouse chromosome 13. Mamm Genome. 1997;8:787-8 pubmed
  62. Lay K, Kume T, Fuchs E. FOXC1 maintains the hair follicle stem cell niche and governs stem cell quiescence to preserve long-term tissue-regenerating potential. Proc Natl Acad Sci U S A. 2016;113:E1506-15 pubmed publisher
    ..Here, we examine this issue by conditionally ablating a key transcription factor Forkhead box C1 (FOXC1) expressed in hair follicle SCs (HFSCs)...
  63. Kaestner K, Lee K, Schlondorff J, Hiemisch H, Monaghan A, Schutz G. Six members of the mouse forkhead gene family are developmentally regulated. Proc Natl Acad Sci U S A. 1993;90:7628-31 pubmed
    ..fkh-1 seems to be the mammalian homologue of the Drosophila FD1 gene, as the sequences are 86% identical. fkh-1 to fkh-6 show distinct spatial patterns of expression in adult tissues and are expressed during embryogenesis. ..
  64. Zaret K. Developmental competence of the gut endoderm: genetic potentiation by GATA and HNF3/fork head proteins. Dev Biol. 1999;209:1-10 pubmed
    ..The mechanistic implications of genetic potentiation and its general significance are discussed. ..
  65. GRUNEBERG H. Exocrine glands and the Chievitz organ of some mouse mutants. J Embryol Exp Morphol. 1971;25:247-61 pubmed
  66. Cesario J, Landin Malt A, Deacon L, Sandberg M, Vogt D, Tang Z, et al. Lhx6 and Lhx8 promote palate development through negative regulation of a cell cycle inhibitor gene, p57Kip2. Hum Mol Genet. 2015;24:5024-39 pubmed publisher
    ..This is the first report elucidating a molecular genetic pathway downstream of Lhx in palate development. ..
  67. Iwao K, Inatani M, Matsumoto Y, Ogata Iwao M, Takihara Y, Irie F, et al. Heparan sulfate deficiency leads to Peters anomaly in mice by disturbing neural crest TGF-beta2 signaling. J Clin Invest. 2009;119:1997-2008 pubmed publisher
    ..This led to reduced phosphorylation of Smad2 and downregulated expression of forkhead box C1 (Foxc1) and paired-like homeodomain transcription factor 2 (Pitx2), transcription factors that have been identified as ..
  68. Pressman C, Chen H, Johnson R. LMX1B, a LIM homeodomain class transcription factor, is necessary for normal development of multiple tissues in the anterior segment of the murine eye. Genesis. 2000;26:15-25 pubmed
    ..In addition, two cDNAs normally downregulated in presumptive cornea, mf1 and mfh1, exhibit persistent expression, while keratocan, a keratin sulfate proteoglycan expressed by keratocytes, ..
  69. Machida A, Okuhara S, Harada K, Iseki S. Difference in apical and basal growth of the frontal bone primordium in Foxc1ch/ch mice. Congenit Anom (Kyoto). 2014;54:172-7 pubmed publisher
    ..A spontaneous loss of Foxc1 function mutant mouse, congenital hydrocephalus (Foxc1(ch/ch)), results in congenital hydrocephalus accompanied by defects in the apical part of the ..
  70. Inman K, Purcell P, Kume T, Trainor P. Interaction between Foxc1 and Fgf8 during mammalian jaw patterning and in the pathogenesis of syngnathia. PLoS Genet. 2013;9:e1003949 pubmed publisher
    ..The etiology and pathogenesis of syngnathia however remains unknown. Here, we report that Foxc1 null embryos display bony syngnathia together with defects in maxillary and mandibular structures, and agenesis of ..
  71. Feiner L, Webber A, Brown C, Lu M, Jia L, Feinstein P, et al. Targeted disruption of semaphorin 3C leads to persistent truncus arteriosus and aortic arch interruption. Development. 2001;128:3061-70 pubmed
    ..Their entry is disrupted in semaphorin 3C mutant mice. These data suggest that semaphorin 3C promotes crest cell migration into the proximal cardiac outflow tract. ..
  72. Komaki F, Miyazaki Y, Niimura F, Matsusaka T, Ichikawa I, Motojima M. Foxc1 gene null mutation causes ectopic budding and kidney hypoplasia but not dysplasia. Cells Tissues Organs. 2013;198:22-7 pubmed publisher
    Mice carrying the null-mutated Foxc1 gene frequently develop an anomalous double collecting system...
  73. Mirzayans F, Lavy R, Penner Chea J, Berry F. Initiation of early osteoblast differentiation events through the direct transcriptional regulation of Msx2 by FOXC1. PLoS ONE. 2012;7:e49095 pubmed publisher
    ..One such factor, the forkhead box transcription factor FOXC1 is necessary for the correct formation of the axial and craniofacial skeleton...
  74. Voss A, Vanyai H, Collin C, Dixon M, McLennan T, Sheikh B, et al. MOZ regulates the Tbx1 locus, and Moz mutation partially phenocopies DiGeorge syndrome. Dev Cell. 2012;23:652-63 pubmed publisher
    ..Our data reveal a molecular mechanism for a specific chromatin modification of the Tbx1 locus intersecting with an environmental determinant, modeling variability in DiGeorge syndrome. ..