Genomes and Genes
Gene Symbol: foxa3
Description: forkhead box A3
Alias: Zffkh1, fa11c04, fkd2, forkhead-2, wu:fa11c04, wu:fc37a08, zf-FKH1, hepatocyte nuclear factor 3-gamma, fc37a08, fork head domain protein 2
- Lopes S, Lourenço R, Pacheco L, Moreno N, Kreiling J, Saude L. Notch signalling regulates left-right asymmetry through ciliary length control. Development. 2010;137:3625-32 pubmed publisher..Notch signalling brings together ciliary length control and fluid flow hydrodynamics with transcriptional activation of laterality genes. In addition, our deltaD mutant analysis discloses an uncoupling between gut and heart laterality. ..
- Gao C, Wang G, Amack J, Mitchell D. Oda16/Wdr69 is essential for axonemal dynein assembly and ciliary motility during zebrafish embryogenesis. Dev Dyn. 2010;239:2190-7 pubmed publisher..Analysis of cilium ultrastructure revealed loss of outer dynein arms in morphant embryos. These results support a remarkable level of functional conservation for Oda16/Wdr69. ..
- Matsui T, Raya A, Kawakami Y, Callol Massot C, Capdevila J, Rodriguez Esteban C, et al. Noncanonical Wnt signaling regulates midline convergence of organ primordia during zebrafish development. Genes Dev. 2005;19:164-75 pubmed..Overall, our results uncover a late, previously unexpected role of noncanonical Wnt signaling in the control of midline assembly of organ precursors during vertebrate embryo development. ..
- Horne Badovinac S, Rebagliati M, Stainier D. A cellular framework for gut-looping morphogenesis in zebrafish. Science. 2003;302:662-5 pubmed..Finally, reducing left-sided Nodal activity randomizes the pattern of LPM migration and gut looping. These results reveal a cellular framework for the regulation of organ laterality by asymmetrically expressed genes. ..
- Sarmah B, Latimer A, Appel B, Wente S. Inositol polyphosphates regulate zebrafish left-right asymmetry. Dev Cell. 2005;9:133-45 pubmed..Our data suggest that the pathway for inositol hexakisphosphate production is a key regulator of asymmetric Ca(2+) flux during LR specification. ..
- Wilkinson C, Carl M, Harris W. Cep70 and Cep131 contribute to ciliogenesis in zebrafish embryos. BMC Cell Biol. 2009;10:17 pubmed publisher..In a number of human diseases, such Bardet-Biedl syndrome, centrosome/cilium proteins are mutated, leading to polycystic kidney disease, situs inversus, and neurological problems, amongst other symptoms...
- Obara T, Mangos S, Liu Y, Zhao J, Wiessner S, Kramer Zucker A, et al. Polycystin-2 immunolocalization and function in zebrafish. J Am Soc Nephrol. 2006;17:2706-18 pubmed
- Goessling W, North T, Lord A, Ceol C, Lee S, Weidinger G, et al. APC mutant zebrafish uncover a changing temporal requirement for wnt signaling in liver development. Dev Biol. 2008;320:161-74 pubmed publisher..These studies reveal an important and time-dependent role for wnt signaling during liver development and regeneration. ..
- Schottenfeld J, Sullivan Brown J, Burdine R. Zebrafish curly up encodes a Pkd2 ortholog that restricts left-side-specific expression of southpaw. Development. 2007;134:1605-15 pubmed..pkd2 also appears to play a role in the propagation of Nodal signals in the LPM. Based on morpholino studies, we propose an additional role for maternal pkd2 in general mesendoderm patterning. ..
- Alexander J, Rothenberg M, Henry G, Stainier D. casanova plays an early and essential role in endoderm formation in zebrafish. Dev Biol. 1999;215:343-57 pubmed..The early endodermal expression of genes such as axial, gata5, and fkd2 does not initiate in casanova mutants, indicating that the endoderm is defective from the onset of gastrulation...
- Panizzi J, Becker Heck A, Castleman V, Al Mutairi D, Liu Y, Loges N, et al. CCDC103 mutations cause primary ciliary dyskinesia by disrupting assembly of ciliary dynein arms. Nat Genet. 2012;44:714-9 pubmed publisher..Chlamydomonas Ccdc103/Pr46b functions as a tightly bound, axoneme-associated protein. These results identify Ccdc103 as a dynein arm attachment factor that causes primary ciliary dyskinesia when mutated. ..
- Serluca F, Xu B, Okabe N, Baker K, Lin S, Sullivan Brown J, et al. Mutations in zebrafish leucine-rich repeat-containing six-like affect cilia motility and result in pronephric cysts, but have variable effects on left-right patterning. Development. 2009;136:1621-31 pubmed publisher..Combined with recently published results, our alleles suggest that the function of seahorse in cilia motility is separable from its function in other cilia-related phenotypes. ..
- Stuckenholz C, Lu L, Thakur P, Kaminski N, Bahary N. FACS-assisted microarray profiling implicates novel genes and pathways in zebrafish gastrointestinal tract development. Gastroenterology. 2009;137:1321-32 pubmed publisher..These experiments identify novel genes and pathways that probably play a role in mammalian gastrointestinal development and are potential targets for therapeutic intervention in the management of gastrointestinal disease and cancer. ..
- Brand M, Heisenberg C, Warga R, Pelegri F, Karlstrom R, Beuchle D, et al. Mutations affecting development of the midline and general body shape during zebrafish embryogenesis. Development. 1996;123:129-42 pubmed..As a working hypothesis, we propose that midline-group genes may act to maintain proper structure and inductive function of zebrafish midline tissues. ..
- Li Z, Korzh V, Gong Z. Localized rbp4 expression in the yolk syncytial layer plays a role in yolk cell extension and early liver development. BMC Dev Biol. 2007;7:117 pubmed..YSL-expressed Rbp4 plays a role in formation of both yolk extension and liver bud, the latter may also require migration of liver progenitor cells. ..
- Yamauchi H, Miyakawa N, Miyake A, Itoh N. Fgf4 is required for left-right patterning of visceral organs in zebrafish. Dev Biol. 2009;332:177-85 pubmed publisher..The present findings indicate that Fgf4 plays a unique role in the LR patterning of visceral organs in zebrafish. ..
- Zhai G, Gu Q, He J, Lou Q, Chen X, Jin X, et al. Sept6 is required for ciliogenesis in Kupffer's vesicle, the pronephros, and the neural tube during early embryonic development. Mol Cell Biol. 2014;34:1310-21 pubmed publisher..Our study reveals a novel role of sept6 in ciliogenesis during early embryonic development in zebrafish. ..
- Carl M, Bianco I, Bajoghli B, Aghaallaei N, Czerny T, Wilson S. Wnt/Axin1/beta-catenin signaling regulates asymmetric nodal activation, elaboration, and concordance of CNS asymmetries. Neuron. 2007;55:393-405 pubmed..We identify a second role for the Wnt pathway in the left/right regulation of LPM Nodal pathway gene expression, and finally, we show that at later stages Axin1 is required for the elaboration of concordant neuroanatomical asymmetries. ..
- Shin D, Shin C, Tucker J, Ober E, Rentzsch F, Poss K, et al. Bmp and Fgf signaling are essential for liver specification in zebrafish. Development. 2007;134:2041-50 pubmed
- Monteiro R, Van Dinther M, Bakkers J, Wilkinson R, Patient R, Ten Dijke P, et al. Two novel type II receptors mediate BMP signalling and are required to establish left-right asymmetry in zebrafish. Dev Biol. 2008;315:55-71 pubmed publisher..We propose a mechanism whereby this differential interpretation of BMP signalling through bmpr2a and bmpr2b is essential for the establishment of LR asymmetry in the zebrafish embryo. ..
- Odenthal J, Nusslein Volhard C. fork head domain genes in zebrafish. Dev Genes Evol. 1998;208:245-58 pubmed..Members of class I (fkd1/axial, fkd2/Zffkh1, fkd4 and fkd7) are differentially transcribed in unsegmented dorsal axial structures such as the floor plate, the ..
- Mayer A, Fishman M. Nil per os encodes a conserved RNA recognition motif protein required for morphogenesis and cytodifferentiation of digestive organs in zebrafish. Development. 2003;130:3917-28 pubmed..Antisense-mediated knockdown of npo results in organ hypoplasia, and overexpression of npo causes an overgrowth of gastrointestinal organs. Thus, npo is a gene essential for a key step in the gut morphogenetic sequence. ..
- Gibert Y, Lattanzi V, Zhen A, Vedder L, Brunet F, Faasse S, et al. BMP signaling modulates hepcidin expression in zebrafish embryos independent of hemojuvelin. PLoS ONE. 2011;6:e14553 pubmed publisher..Taken together, these data support an alternative mechanism for hepcidin regulation during zebrafish embryonic development, which is independent of hjv. ..
- Hong S, Dawid I. FGF-dependent left-right asymmetry patterning in zebrafish is mediated by Ier2 and Fibp1. Proc Natl Acad Sci U S A. 2009;106:2230-5 pubmed publisher..We conclude that Ier2 and Fibp1 mediate FGF signaling in ciliogenesis in Kupffer's Vesicle and in the establishment of laterality in the zebrafish embryo. ..
- Hammerschmidt M, Pelegri F, Mullins M, Kane D, Brand M, van Eeden F, et al. Mutations affecting morphogenesis during gastrulation and tail formation in the zebrafish, Danio rerio. Development. 1996;123:143-51 pubmed..Mutants in kugelig (kgg) do not form the yolk tube at the posterior side of the yolk sac. ..
- Chen J, van Bebber F, Goldstein A, Serluca F, Jackson D, Childs S, et al. Genetic steps to organ laterality in zebrafish. Comp Funct Genomics. 2001;2:60-8 pubmed publisher..That laterality decisions of organs may be independently perturbed, as in the Class 3 mutation, indicates that there are distinctive pathways for reception and organotypic interpretation of the global signals. ..
- Warga R, Nusslein Volhard C. Origin and development of the zebrafish endoderm. Development. 1999;126:827-38 pubmed..This suggests that a common pathway initially specifies germ layers together before a progressive sequence of determinative events segregate endoderm and mesoderm into morphologically distinct germ layers. ..
- Rodaway A, Takeda H, Koshida S, Broadbent J, Price B, Smith J, et al. Induction of the mesendoderm in the zebrafish germ ring by yolk cell-derived TGF-beta family signals and discrimination of mesoderm and endoderm by FGF. Development. 1999;126:3067-78 pubmed
- Dubrulle J, Jordan B, Akhmetova L, Farrell J, Kim S, Solnica Krezel L, et al. Response to Nodal morphogen gradient is determined by the kinetics of target gene induction. elife. 2015;4: pubmed publisher..Thus, the timing and magnitude of target gene expression can be used to modulate the range of expression and diversify the response to morphogen gradients. ..
- Jaffe K, Grimes D, Schottenfeld Roames J, Werner M, Ku T, Kim S, et al. c21orf59/kurly Controls Both Cilia Motility and Polarization. Cell Rep. 2016;14:1841-9 pubmed publisher..Kur also makes interactions with other PCP components, including Disheveled. This supports a model wherein Kur plays a dual role in cilia motility and polarization. ..
- Fuller K, O Connell J, Gordon J, Mauti O, EGGENSCHWILER J. Rab23 regulates Nodal signaling in vertebrate left-right patterning independently of the Hedgehog pathway. Dev Biol. 2014;391:182-95 pubmed publisher..Collectively, these data suggest that Rab23 is an essential component of the mechanism that transmits asymmetric patterning information from the node to the LPM. ..
- Deshwar A, Chng S, Ho L, Reversade B, Scott I. The Apelin receptor enhances Nodal/TGF? signaling to ensure proper cardiac development. elife. 2016;5: pubmed publisher..Our results favour a model in which Aplnr is required to fine-tune Nodal output, acting as a specific rheostat for the Nodal/TGF? pathway during the earliest stages of cardiogenesis. ..
- Leung T, Humbert J, Stauffer A, Giger K, Chen H, Tsai H, et al. The orphan G protein-coupled receptor 161 is required for left-right patterning. Dev Biol. 2008;323:31-40 pubmed publisher..Taken together, these data support a model in which gpr161 plays an essential role in left-right (L-R) patterning by modulating Ca(2+) levels in the cells surrounding the Kupffer's vesicle. ..
- Serluca F, Drummond I, Fishman M. Endothelial signaling in kidney morphogenesis: a role for hemodynamic forces. Curr Biol. 2002;12:492-7 pubmed..Thus, vascular flow is required for glomerular assembly, most probably acting via a stretch-responsive signaling system in the vessel wall. ..
- Teoh P, Shu Chien A, Chan W. Pbx1 is essential for growth of zebrafish swim bladder. Dev Dyn. 2010;239:865-74 pubmed publisher..The knockdown of pbx1 did not perturb the expression of prdc and foxA3, with both early swim bladder markers appearing normally at 36 and 48 hpf, respectively...
- Kim Y, Epting D, Slanchev K, Engel C, Walz G, Kramer Zucker A. A complex of BBS1 and NPHP7 is required for cilia motility in zebrafish. PLoS ONE. 2013;8:e72549 pubmed publisher..2 did not compromise cilia formation, but disrupted cilia motility. Although NPHP7 has been shown to act as transcriptional repressor, our studies suggest a crosstalk between BBS1 and NPHP7 in regulating normal function of the cilium. ..
- Wang G, Cadwallader A, Jang D, Tsang M, Yost H, Amack J. The Rho kinase Rock2b establishes anteroposterior asymmetry of the ciliated Kupffer's vesicle in zebrafish. Development. 2011;138:45-54 pubmed publisher..Our results suggest a link between AP patterning of the ciliated Kupffer's vesicle and LR patterning of the zebrafish embryo. ..
- Porreca I, De Felice E, Fagman H, Di Lauro R, Sordino P. Zebrafish bcl2l is a survival factor in thyroid development. Dev Biol. 2012;366:142-52 pubmed publisher..This is the first demonstration of an active mechanism to ensure survival of the thyroid primordium during morphogenesis. ..
- Neugebauer J, Cadwallader A, Amack J, Bisgrove B, Yost H. Differential roles for 3-OSTs in the regulation of cilia length and motility. Development. 2013;140:3892-902 pubmed publisher..We propose that individual 3-OST isozymes create distinct modified domains or 'glycocodes' on cell surface proteoglycans, which in turn regulate the response to diverse cell signaling pathways. ..
- Wu J, Choi T, Shin D. tomm22 Knockdown-Mediated Hepatocyte Damages Elicit Both the Formation of Hybrid Hepatocytes and Biliary Conversion to Hepatocytes in Zebrafish Larvae. Gene Expr. 2017;17:237-249 pubmed publisher..This new liver injury model in which both hepatocytes and BECs contribute to regenerated hepatocytes will aid in better understanding the mechanisms of innate liver regeneration in the diseased liver. ..
- Shin D, Weidinger G, Moon R, Stainier D. Intrinsic and extrinsic modifiers of the regulative capacity of the developing liver. Mech Dev. 2012;128:525-35 pubmed publisher..Altogether, these studies reveal that there is more than one way to form a liver, and provide molecular insights into the phenomenon of tissue plasticity. ..
- Reichenbach B, Delalande J, Kolmogorova E, Prier A, Nguyen T, Smith C, et al. Endoderm-derived Sonic hedgehog and mesoderm Hand2 expression are required for enteric nervous system development in zebrafish. Dev Biol. 2008;318:52-64 pubmed publisher..Furthermore we show that endoderm and HH signaling, but not hand2, regulate gdnf expression in the intestine, highlighting a central role of endoderm and SHH in patterning the intestine and the ENS. ..
- Song J, Kim H, Gong Z, Liu N, Lin S. Vhnf1 acts downstream of Bmp, Fgf, and RA signals to regulate endocrine beta cell development in zebrafish. Dev Biol. 2007;303:561-75 pubmed..Taken together, our data establish that vhnf1 acts downstream of the signaling pathways of RA, Bmp, and Fgf to regulate pancreas development in zebrafish. ..
- Lee S, So J, Kim H, Choi J, Lee M, Choi S, et al. Angiopoietin-like 3 regulates hepatocyte proliferation and lipid metabolism in zebrafish. Biochem Biophys Res Commun. 2014;446:1237-42 pubmed publisher..Together, our findings propose a novel role for Angptl3 in liver cell proliferation and maintenance during zebrafish embryogenesis. Finally, angptl3 morphants will serve as a good model for understanding the pathophysiology of FHBL2. ..
- Burkhalter M, Fralish G, Premont R, Caron M, Philipp M. Grk5l controls heart development by limiting mTOR signaling during symmetry breaking. Cell Rep. 2013;4:625-32 pubmed publisher..These findings could implicate GRK5 as a susceptibility allele for certain cases of CHD. ..
- Müller I, Knapik E, Hatzopoulos A. Expression of the protein related to Dan and Cerberus gene--prdc--During eye, pharyngeal arch, somite, and swim bladder development in zebrafish. Dev Dyn. 2006;235:2881-8 pubmed..Moreover, the complexity of the prdc expression pattern suggests possible roles in eye development, pharyngeal arch remodeling, somitogenesis, and swim bladder organogenesis...
- Lahvic J, Ji Y, Marín P, Zuflacht J, Springel M, Wosen J, et al. Small heat shock proteins are necessary for heart migration and laterality determination in zebrafish. Dev Biol. 2013;384:166-80 pubmed publisher..In embryos in which the reduction of hspb7 or hspb12 was limited to the yolk, migration defects predominated, suggesting that the yolk expression of these genes rather than heart expression is responsible for the migration defects. ..
- Li Y, Farooq M, Sheng D, Chandramouli C, Lan T, Mahajan N, et al. Augmenter of liver regeneration (alr) promotes liver outgrowth during zebrafish hepatogenesis. PLoS ONE. 2012;7:e30835 pubmed publisher..The dose-dependent and partial suppression of alr expression through MO-mediated knockdown allows the identification of its late developmental role in vertebrate liver organogenesis. ..
- Lu G, Ren S, Korge P, Choi J, Dong Y, Weiss J, et al. A novel mitochondrial matrix serine/threonine protein phosphatase regulates the mitochondria permeability transition pore and is essential for cellular survival and development. Genes Dev. 2007;21:784-96 pubmed..These data suggest that PP2Cm is a novel mitochondrial protein phosphatase that has a critical function in cell death and survival, and may play a role in regulating the MPTP opening. ..
- Yaklichkin S, Vekker A, Stayrook S, Lewis M, Kessler D. Prevalence of the EH1 Groucho interaction motif in the metazoan Fox family of transcriptional regulators. BMC Genomics. 2007;8:201 pubmed
- Li Y, Yagi H, Onuoha E, Damerla R, Francis R, Furutani Y, et al. DNAH6 and Its Interactions with PCD Genes in Heterotaxy and Primary Ciliary Dyskinesia. PLoS Genet. 2016;12:e1005821 pubmed publisher..Together, these findings support an oligogenic disease model with broad relevance for further interrogating the genetic etiology of human ciliopathies. ..
- Noël E, Verhoeven M, Lagendijk A, Tessadori F, Smith K, Choorapoikayil S, et al. A Nodal-independent and tissue-intrinsic mechanism controls heart-looping chirality. Nat Commun. 2013;4:2754 pubmed publisher..We find that Nodal signalling regulates actin gene expression, supporting a model in which Nodal signalling amplifies this tissue-intrinsic mechanism of heart looping. ..
- Smith K, Lagendijk A, Courtney A, Chen H, Paterson S, Hogan B, et al. Transmembrane protein 2 (Tmem2) is required to regionally restrict atrioventricular canal boundary and endocardial cushion development. Development. 2011;138:4193-8 pubmed publisher..Finally, we show that immature AVC expansion in wkm mutants is rescued by depleting Bmp4, indicating that Tmem2 restricts bmp4 expression to delimit the AVC primordium during cardiac development. ..
- Verleyen D, Luyten F, Tylzanowski P. Orphan G-protein coupled receptor 22 (Gpr22) regulates cilia length and structure in the zebrafish Kupffer's vesicle. PLoS ONE. 2014;9:e110484 pubmed publisher..Further, we found that Gpr22 does not act upstream of the two cilia master regulators, Foxj1a and Rfx2. To conclude, our study characterized a novel player in the field of ciliogenesis. ..
- Schueler M, Braun D, Chandrasekar G, Gee H, Klasson T, Halbritter J, et al. DCDC2 mutations cause a renal-hepatic ciliopathy by disrupting Wnt signaling. Am J Hum Genet. 2015;96:81-92 pubmed publisher..We thus demonstrate a central role of Wnt signaling in the pathogenesis of NPHP-RC, suggesting an avenue for potential treatment of NPHP-RC. ..
- Levic D, Minkel J, Wang W, Rybski W, Melville D, Knapik E. Animal model of Sar1b deficiency presents lipid absorption deficits similar to Anderson disease. J Mol Med (Berl). 2015;93:165-76 pubmed publisher..Key messages: Sar1b depletion phenotype in zebrafish resembles Anderson disease deficits. Sar1b deficiency results in multi-organ developmental deficits. Sar1b is required for dietary cholesterol uptake into enterocytes. ..
- Fukuda K, Kikuchi Y. Endoderm development in vertebrates: fate mapping, induction and regional specification. Dev Growth Differ. 2005;47:343-55 pubmed..We discuss the classical fate mapping of the endoderm and the more recent progress in characterizing its induction, segregation and regional specification. ..
- Pintado P, Sampaio P, Tavares B, Montenegro Johnson T, Smith D, Lopes S. Dynamics of cilia length in left-right development. R Soc Open Sci. 2017;4:161102 pubmed publisher..This mild reduction is corroborated by experiments, providing a mechanism for the mild impact on organ situs. ..
- Ko S, Choi T, Russell J, So J, Monga S, Shin D. Bromodomain and extraterminal (BET) proteins regulate biliary-driven liver regeneration. J Hepatol. 2016;64:316-325 pubmed publisher..BET proteins regulate BEC-driven liver regeneration at multiple steps: BEC dedifferentiation, HB-LC proliferation, the proliferation of newly generated hepatocytes, and hepatocyte maturation. ..
- Zhen A, Nguyen N, Gibert Y, Motola S, Buckett P, Wessling Resnick M, et al. The small molecule, genistein, increases hepcidin expression in human hepatocytes. Hepatology. 2013;58:1315-25 pubmed publisher..Genistein and other candidate molecules may subsequently be developed into new therapies for iron overload syndromes...
- Fogelgren B, Lin S, Zuo X, Jaffe K, Park K, Reichert R, et al. The exocyst protein Sec10 interacts with Polycystin-2 and knockdown causes PKD-phenotypes. PLoS Genet. 2011;7:e1001361 pubmed publisher..Our work supports a model in which the exocyst is required for the ciliary localization of polycystin-2, thus allowing for polycystin-2 function in cellular processes. ..
- Kopinke D, Sasine J, Swift J, Stephens W, Piotrowski T. Retinoic acid is required for endodermal pouch morphogenesis and not for pharyngeal endoderm specification. Dev Dyn. 2006;235:2695-709 pubmed..These results demonstrate that the developmental processes underlying pharyngeal arch defects differ depending on when RA signaling is disturbed during development...
- Lai Y, Lu Y, Lien H, Huang C, Hwang S. Foxa2 and Hif1ab regulate maturation of intestinal goblet cells by modulating agr2 expression in zebrafish embryos. Biochem J. 2016;473:2205-18 pubmed publisher..These results demonstrate that Foxa1 regulates agr2 expression in the pharynx, whereas both Foxa2 and Hif1ab control agr2 expression in intestinal goblet cells to regulate maturation of these cells. ..
- Shu X, Huang J, Dong Y, Choi J, Langenbacher A, Chen J. Na,K-ATPase alpha2 and Ncx4a regulate zebrafish left-right patterning. Development. 2007;134:1921-30 pubmed