Genomes and Genes
Gene Symbol: pax6a
Description: paired box 6a
Alias: PAXZF-A, Pax6.1, cb280, fc20e07, pax-a, pax6, pax[zf-a], paxzfa, wu:fc20e07, zfpax-6a, zfpax-6b, paired box protein Pax-6, etID309716.25, paired box gene 6a, paired box homeotic gene 6, paired box protein Pax[Zf-a], pax-6
- Guner B, Karlstrom R. Cloning of zebrafish nkx6.2 and a comprehensive analysis of the conserved transcriptional response to Hedgehog/Gli signaling in the zebrafish neural tube. Gene Expr Patterns. 2007;7:596-605 pubmed
- Kikuta H, Laplante M, Navratilova P, Komisarczuk A, Engström P, Fredman D, et al. Genomic regulatory blocks encompass multiple neighboring genes and maintain conserved synteny in vertebrates. Genome Res. 2007;17:545-55 pubmed..Reporter insertions distal to zebrafish developmental regulatory genes pax6.1/2, rx3, id1, and fgf8 and miRNA genes mirn9-1 and mirn9-5 recapitulate the expression patterns of these genes ..
- Chandrasekar G, Lauter G, Hauptmann G. Distribution of corticotropin-releasing hormone in the developing zebrafish brain. J Comp Neurol. 2007;505:337-51 pubmed..The widespread distribution of CRH-synthesizing cells outside the preoptic region suggests additional functions of CRH in the embryonic zebrafish brain. ..
- Fausett B, Gumerson J, Goldman D. The proneural basic helix-loop-helix gene ascl1a is required for retina regeneration. J Neurosci. 2008;28:1109-17 pubmed publisher..that in zebrafish, retinal injury stimulates Müller glia to generate multipotent alpha1-tubulin (alpha1T) and pax6-expressing progenitors for retinal repair...
- Scholpp S, Lohs C, Brand M. Engrailed and Fgf8 act synergistically to maintain the boundary between diencephalon and mesencephalon. Development. 2003;130:4881-93 pubmed..Expression of pax6.1, a key regulator of forebrain development, is posteriorly suppressed by the Engrailed proteins, Eng2 and Eng3...
- Walshe J, Mason I. Unique and combinatorial functions of Fgf3 and Fgf8 during zebrafish forebrain development. Development. 2003;130:4337-49 pubmed..Analysis of embryos treated with an FGFR inhibitor suggests that continuous FGF signalling is required from gastrulation stages for normal forebrain patterning, and identifies additional requirements for FGFR activity. ..
- Macdonald R, Xu Q, Barth K, Mikkola I, Holder N, Fjose A, et al. Regulatory gene expression boundaries demarcate sites of neuronal differentiation in the embryonic zebrafish forebrain. Neuron. 1994;13:1039-53 pubmed..This strip of cells fails to develop in mutant fish in which specification of the ventral CNS is disrupted, suggesting that its development may be regulated by the same inductive pathways that pattern the ventral midline. ..
- Heisenberg C, Brennan C, Wilson S. Zebrafish aussicht mutant embryos exhibit widespread overexpression of ace (fgf8) and coincident defects in CNS development. Development. 1999;126:2129-40 pubmed..We conclude that aus represents a mutation involving a locus normally required for the regulation of ace expression during embryogenesis. ..
- Sekimizu K, Nishioka N, Sasaki H, Takeda H, Karlstrom R, Kawakami A. The zebrafish iguana locus encodes Dzip1, a novel zinc-finger protein required for proper regulation of Hedgehog signaling. Development. 2004;131:2521-32 pubmed..Taken together, our studies show that Igu/Dzip1 functions as a permissive factor that is required for the proper regulation of Hh target genes in response to Hh signals. ..
- Kawakami A, Nojima Y, Toyoda A, Takahoko M, Satoh M, Tanaka H, et al. The zebrafish-secreted matrix protein you/scube2 is implicated in long-range regulation of hedgehog signaling. Curr Biol. 2005;15:480-8 pubmed..We further show that Bmp activity can be attenuated by the coexpression of Scube2. Our data support the idea that Scube2 can modulate the long-range action of Bmp-dependent signaling in the neural tube and somites. ..
- Miyake A, Nakayama Y, Konishi M, Itoh N. Fgf19 regulated by Hh signaling is required for zebrafish forebrain development. Dev Biol. 2005;288:259-75 pubmed..The present findings indicate that Fgf19 signaling is crucial for forebrain development by interacting with Hh and provide new insights into the roles of Fgf signaling in brain development. ..
- Seiliez I, Thisse B, Thisse C. FoxA3 and goosecoid promote anterior neural fate through inhibition of Wnt8a activity before the onset of gastrulation. Dev Biol. 2006;290:152-63 pubmed..Altogether, foxA3 and goosecoid cooperate to promote formation of anterior neural tissue by protecting, as early as blastula stage, presumptive anterior neural cells from an irreversible caudalization by the posteriorizing factor Wnt8a. ..
- Sherpa T, Fimbel S, Mallory D, Maaswinkel H, Spritzer S, Sand J, et al. Ganglion cell regeneration following whole-retina destruction in zebrafish. Dev Neurobiol. 2008;68:166-81 pubmed..This model system for whole-retina regeneration has provided an informative view of the regenerative process. ..
- Scholpp S, Brand M. Integrity of the midbrain region is required to maintain the diencephalic-mesencephalic boundary in zebrafish no isthmus/pax2.1 mutants. Dev Dyn. 2003;228:313-22 pubmed..We therefore suggest that the genetic program controlled by Pax2.1 is not only involved in initiating but also in maintaining the identity of midbrain and isthmus cells to prevent them from assuming a forebrain or hindbrain fate. ..
- Plessy C, Dickmeis T, Chalmel F, Strahle U. Enhancer sequence conservation between vertebrates is favoured in developmental regulator genes. Trends Genet. 2005;21:207-10 pubmed
- Mikkola I, Bruun J, Holm T, Johansen T. Superactivation of Pax6-mediated transactivation from paired domain-binding sites by dna-independent recruitment of different homeodomain proteins. J Biol Chem. 2001;276:4109-18 pubmedThe Pax6 genes encode evolutionary conserved transcription factors that act high up in the regulatory hierarchy controlling development of central organs such as the eyes and the central nervous system...
- Navratilova P, Fredman D, Hawkins T, Turner K, Lenhard B, Becker T. Systematic human/zebrafish comparative identification of cis-regulatory activity around vertebrate developmental transcription factor genes. Dev Biol. 2009;327:526-40 pubmed publisher..On the loci of two developmental transcription factor genes, SOX3 and PAX6, we demonstrate that HCNEs conserved between human and zebrafish can be systematically and reliably tested for ..
- Kim S, Shin J, Park H, Yeo S, Hong S, Han S, et al. Specification of an anterior neuroectoderm patterning by Frizzled8a-mediated Wnt8b signalling during late gastrulation in zebrafish. Development. 2002;129:4443-55 pubmed..In conclusion, we suggest that a gradient of Fz8a-mediated Wnt8b signalling may play crucial role in patterning the posterior diencephalon and midbrain regions in the late gastrula. ..
- Kashyap B, Frederickson L, Stenkamp D. Mechanisms for persistent microphthalmia following ethanol exposure during retinal neurogenesis in zebrafish embryos. Vis Neurosci. 2007;24:409-21 pubmed..We conclude that ethanol exposure over the time of retinal neurogenesis resulted in persistent microphthalmia due to a combination of an overall developmental delay, lens abnormalities, and reduced retinal cell differentiation. ..
- Sanek N, Taylor A, Nyholm M, Grinblat Y. Zebrafish zic2a patterns the forebrain through modulation of Hedgehog-activated gene expression. Development. 2009;136:3791-800 pubmed publisher..These data uncover a novel, essential role for Zic2a as a modulator of Hh-activated gene expression in the developing forebrain and advance our understanding of a key gene regulatory network that, when disrupted, causes HPE. ..
- Lakowski J, Majumder A, Lauderdale J. Mechanisms controlling Pax6 isoform expression in the retina have been conserved between teleosts and mammals. Dev Biol. 2007;307:498-520 pubmed..Using bacterial artificial chromosome (BAC) technology, we modified a zebrafish Pax6a BAC such that we could distinguish paired-containing Pax6a transcripts from paired-less Pax6a transcripts...
- Ekker S, Ungar A, Greenstein P, von Kessler D, Porter J, Moon R, et al. Patterning activities of vertebrate hedgehog proteins in the developing eye and brain. Curr Biol. 1995;5:944-55 pubmed
- Tobin J, Di Franco M, Eichers E, May Simera H, Garcia M, Yan J, et al. Inhibition of neural crest migration underlies craniofacial dysmorphology and Hirschsprung's disease in Bardet-Biedl syndrome. Proc Natl Acad Sci U S A. 2008;105:6714-9 pubmed publisher..Moreover, this is a previously undescribed method of using characterization of facial dysmorphology as a basis for investigating the pathomechanism of CF development in dysmorphic syndromes...
- Buckles G, Thorpe C, Ramel M, Lekven A. Combinatorial Wnt control of zebrafish midbrain-hindbrain boundary formation. Mech Dev. 2004;121:437-47 pubmed
- Delporte F, Pasque V, Devos N, Manfroid I, Voz M, Motte P, et al. Expression of zebrafish pax6b in pancreas is regulated by two enhancers containing highly conserved cis-elements bound by PDX1, PBX and PREP factors. BMC Dev Biol. 2008;8:53 pubmed publisher..Due to genome duplication in the evolution of ray-finned fishes, zebrafish has two pax6 genes, pax6a and pax6b...
- Scholpp S, Foucher I, Staudt N, Peukert D, Lumsden A, Houart C. Otx1l, Otx2 and Irx1b establish and position the ZLI in the diencephalon. Development. 2007;134:3167-76 pubmed..We therefore propose that the ZLI is induced within the competence area established by Otx1l/2, and is posteriorly restricted by Irx1b. ..
- Loosli F, Staub W, Finger Baier K, Ober E, Verkade H, Wittbrodt J, et al. Loss of eyes in zebrafish caused by mutation of chokh/rx3. EMBO Rep. 2003;4:894-9 pubmed..Thus, these mutations in orthologous genes allow us to study the evolution of vertebrate eye development at the molecular level. ..
- Varga Z, Wegner J, Westerfield M. Anterior movement of ventral diencephalic precursors separates the primordial eye field in the neural plate and requires cyclops. Development. 1999;126:5533-46 pubmed..Our results indicate that movement of a median subpopulation of diencephalic precursors separates retinal precursors into left and right eyes. Wild-type cyclops gene function is required for these morphogenetic movements. ..
- Blader P, Lam C, Rastegar S, Scardigli R, Nicod J, Simplicio N, et al. Conserved and acquired features of neurogenin1 regulation. Development. 2004;131:5627-37 pubmed..This regulatory region, which drives expression in the zebrafish diencephalon and hindbrain, is dependent on Pax6 activity and binds recombinant Pax6 in vitro...
- Kleinjan D, Bancewicz R, Gautier P, Dahm R, Schonthaler H, Damante G, et al. Subfunctionalization of duplicated zebrafish pax6 genes by cis-regulatory divergence. PLoS Genet. 2008;4:e29 pubmed publisher..In zebrafish, following a postulated whole-genome duplication event in an ancestral teleost, duplicates pax6a and pax6b jointly fulfill these roles...
- Thummel R, Kassen S, Montgomery J, Enright J, Hyde D. Inhibition of Müller glial cell division blocks regeneration of the light-damaged zebrafish retina. Dev Neurobiol. 2008;68:392-408 pubmed..to starting intense light exposure inhibited both Müller glial cell proliferation and neuronal progenitor marker Pax6 expression...
- Hauptmann G, Söll I, Gerster T. The early embryonic zebrafish forebrain is subdivided into molecularly distinct transverse and longitudinal domains. Brain Res Bull. 2002;57:371-5 pubmed..Furthermore, we identified a series of eight transverse diencephalic domains which may indicate a prosomeric organization of the rostral zebrafish brain. ..
- Kim J, Lauderdale J. Analysis of Pax6 expression using a BAC transgene reveals the presence of a paired-less isoform of Pax6 in the eye and olfactory bulb. Dev Biol. 2006;292:486-505 pubmedb>Pax6, a member of the paired-family of transcription factors, exhibits restricted expression and essential functions in the developing eye, olfactory system, central nervous system, and pancreas...
- Macdonald R, Barth K, Xu Q, Holder N, Mikkola I, Wilson S. Midline signalling is required for Pax gene regulation and patterning of the eyes. Development. 1995;121:3267-78 pubmedb>Pax6 and Pax2 are members of the Pax family of transcription factors that are both expressed in the developing visual system of zebrafish embryos...
- Zhang C, Turton Q, Mackinnon S, Sulik K, Cole G. Agrin function associated with ocular development is a target of ethanol exposure in embryonic zebrafish. Birth Defects Res A Clin Mol Teratol. 2011;91:129-41 pubmed publisher..of agrin MO and ethanol produces pronounced microphthalmia, markedly reduces agrin gene expression, and perturbs Pax6a and Mbx gene expression...
- Lee J, Willer J, Willer G, Smith K, Gregg R, Gross J. Zebrafish blowout provides genetic evidence for Patched1-mediated negative regulation of Hedgehog signaling within the proximal optic vesicle of the vertebrate eye. Dev Biol. 2008;319:10-22 pubmed publisher..of the proximal optic vesicle marker pax2a is expanded in blowout at the expense of the distal optic vesicle marker pax6, suggesting that the initial patterning of the optic vesicle into proximal and distal territories is disrupted in ..
- Zhang C, Frazier J, Chen H, Liu Y, Lee J, Cole G. Molecular and morphological changes in zebrafish following transient ethanol exposure during defined developmental stages. Neurotoxicol Teratol. 2014;44:70-80 pubmed publisher..depends on the embryonic stage and concentration of alcohol exposure, as does diminution of retinal Pax6a or forebrain and hindbrain GAD1 gene expression...
- Ando H, Kobayashi M, Tsubokawa T, Uyemura K, Furuta T, Okamoto H. Lhx2 mediates the activity of Six3 in zebrafish forebrain growth. Dev Biol. 2005;287:456-68 pubmed..Our results suggest that Lhx2 may mediate an alternative or parallel pathway for control of cellular proliferation in the developing forebrain via Six3. ..
- Tallafuss A, Bally Cuif L. Tracing of her5 progeny in zebrafish transgenics reveals the dynamics of midbrain-hindbrain neurogenesis and maintenance. Development. 2003;130:4307-23 pubmed..Together, our results suggest a model where an ordered MH pro-domain is identified at gastrulation, and where cell identity choices within this domain are subsequently differentially controlled by Fgf8 and Pax2.1 functions. ..
- Ravi V, Bhatia S, Gautier P, Loosli F, Tay B, Tay A, et al. Sequencing of Pax6 loci from the elephant shark reveals a family of Pax6 genes in vertebrate genomes, forged by ancient duplications and divergences. PLoS Genet. 2013;9:e1003177 pubmed publisherb>Pax6 is a developmental control gene essential for eye development throughout the animal kingdom. In addition, Pax6 plays key roles in other parts of the CNS, olfactory system, and pancreas...
- Kassen S, Ramanan V, Montgomery J, T Burket C, Liu C, Vihtelic T, et al. Time course analysis of gene expression during light-induced photoreceptor cell death and regeneration in albino zebrafish. Dev Neurobiol. 2007;67:1009-31 pubmed..Some of the Stat3-positive Müller cells expressed PCNA at 31 h, suggesting that Stat3 may play a role in signaling a subset of Müller cells to proliferate during the regeneration response. ..
- Fausett B, Goldman D. A role for alpha1 tubulin-expressing Müller glia in regeneration of the injured zebrafish retina. J Neurosci. 2006;26:6303-13 pubmed..These data strongly suggest alpha1T-expressing Müller glia dedifferentiate and mediate regeneration of the injured zebrafish retina. ..
- Peng G, Westerfield M. Lhx5 promotes forebrain development and activates transcription of secreted Wnt antagonists. Development. 2006;133:3191-200 pubmed..Our results demonstrate that Lhx5 is a required factor that promotes forebrain development and inhibits Wnt signaling by activating the transcription of secreted Wnt antagonists. ..
- Nolte C, Rastegar M, Amores A, Bouchard M, Grote D, Maas R, et al. Stereospecificity and PAX6 function direct Hoxd4 neural enhancer activity along the antero-posterior axis. Dev Biol. 2006;299:582-93 pubmed..Additionally, stage-matched zebrafish embryos having decreased pax6a and/or pax6b activity display malformed rhombomere boundaries and an anteriorized hoxd4a expression border...
- Hauptmann G, Gerster T. Regulatory gene expression patterns reveal transverse and longitudinal subdivisions of the embryonic zebrafish forebrain. Mech Dev. 2000;91:105-18 pubmed..Our data suggest a strong conservation of early forebrain organization between lower and higher vertebrates. ..
- Peukert D, Weber S, Lumsden A, Scholpp S. Lhx2 and Lhx9 determine neuronal differentiation and compartition in the caudal forebrain by regulating Wnt signaling. PLoS Biol. 2011;9:e1001218 pubmed publisher..We therefore suggest that after initial neural tube patterning, neurogenesis within a brain compartment influences the integrity of the neuronal progenitor pool and border formation of a neuromeric compartment. ..
- Gonzalez Nunez V. Role of gabra2, GABAA receptor alpha-2 subunit, in CNS development. Biochem Biophys Rep. 2015;3:190-201 pubmed publisher..system was also affected in gabra2 morpholino-injected embryos, although no changes were found for pax6a and shh a expression...
- Zhao X, Kuja Panula J, Rouhiainen A, Chen Y, Panula P, Rauvala H. High mobility group box-1 (HMGB1; amphoterin) is required for zebrafish brain development. J Biol Chem. 2011;286:23200-13 pubmed publisher..and proliferation of neural progenitors and displays fewer cell groups expressing the transcription factor Pax6a in the forebrain and aberrant Wnt8 signaling...
- Li Y, Li G, Wang H, Du J, Yan J. Analysis of a gene regulatory cascade mediating circadian rhythm in zebrafish. PLoS Comput Biol. 2013;9:e1002940 pubmed publisher..Our study describes a systematic method to discover previously unidentified TFs involved in circadian physiology in complex organisms. ..
- Wargelius A, Seo H, Austbø L, Fjose A. Retinal expression of zebrafish six3.1 and its regulation by Pax6. Biochem Biophys Res Commun. 2003;309:475-81 pubmed..This analysis also revealed specific binding of zebrafish Pax6.1 protein to an element required for six3.1 expression in ganglion cells. Furthermore, an enhancement of six3...
- Han R, Wang R, Zhao Q, Han Y, Zong S, Miao S, et al. Trim69 regulates zebrafish brain development by ap-1 pathway. Sci Rep. 2016;6:24034 pubmed publisher..Overall, our results support a role for trim69 in the development of the zebrafish brain through ap-1 pathway. ..
- Clark A, Yun S, Veien E, Wu Y, Chow R, Dorsky R, et al. Negative regulation of Vsx1 by its paralog Chx10/Vsx2 is conserved in the vertebrate retina. Brain Res. 2008;1192:99-113 pubmed
- Amirthalingam K, Lorens J, Saetre B, Salaneck E, Fjose A. Embryonic expression and DNA-binding properties of zebrafish pax-6. Biochem Biophys Res Commun. 1995;215:122-8 pubmed..The relative DNA-binding affinity of the zebrafish Pax-6 protein to different categories of Pax recognition sites is shared with the murine homologue. ..
- Kaslin J, Ganz J, Geffarth M, Grandel H, Hans S, Brand M. Stem cells in the adult zebrafish cerebellum: initiation and maintenance of a novel stem cell niche. J Neurosci. 2009;29:6142-53 pubmed publisher..Nevertheless, retained epithelial properties such as distinct polarization and ventricular contact are critical common determinants to maintain neural stem cell activity in vertebrates. ..
- Lele Z, Folchert A, Concha M, Rauch G, Geisler R, Rosa F, et al. parachute/n-cadherin is required for morphogenesis and maintained integrity of the zebrafish neural tube. Development. 2002;129:3281-94 pubmed..Our results thus highlight novel and crucial in vivo roles for Ncad in the control of cell convergence, maintenance of neuronal positioning and dorsal cell proliferation during vertebrate neural tube development. ..
- Bhatia S, Monahan J, Ravi V, Gautier P, Murdoch E, Brenner S, et al. A survey of ancient conserved non-coding elements in the PAX6 locus reveals a landscape of interdigitated cis-regulatory archipelagos. Dev Biol. 2014;387:214-28 pubmed publisher..with transgenic mouse and zebrafish reporter assays to survey the genomic locus of the developmental control gene PAX6 for the presence of novel cis-regulatory elements...
- Stanganello E, Hagemann A, Mattes B, Sinner C, Meyen D, Weber S, et al. Filopodia-based Wnt transport during vertebrate tissue patterning. Nat Commun. 2015;6:5846 pubmed publisher..Indeed, we show that a filopodia-based transport system for Wnt8a controls anteroposterior patterning of the neural plate during vertebrate gastrulation. ..
- Lin C, Huang H, Lu P, Lin C, Lu K, Tsai H. Ras-Related Nuclear Protein is required for late developmental stages of retinal cells in zebrafish eyes. Int J Dev Biol. 2015;59:435-42 pubmed publisher..Early eye field specification markers, rx1 and pax6, were only slightly affected, and markers for establishing axon migration, fgf8 and pax2, were normally expressed, ..
- Chaplin N, Tendeng C, Wingate R. Absence of an external germinal layer in zebrafish and shark reveals a distinct, anamniote ground plan of cerebellum development. J Neurosci. 2010;30:3048-57 pubmed publisher..We show that these anamniotes share a common ground plan of granule cell production that does not include an external germinal layer. ..