gdf6a

Summary

Gene Symbol: gdf6a
Description: growth differentiation factor 6a
Alias: rdr, sb:cb278, growth/differentiation factor 6-A, GDF-6-A, cb278, darl, growth differentiation factor 6, out, protein radar, radar
Species: zebrafish
Products:     gdf6a

Top Publications

  1. Gosse N, Baier H. An essential role for Radar (Gdf6a) in inducing dorsal fate in the zebrafish retina. Proc Natl Acad Sci U S A. 2009;106:2236-41 pubmed publisher
    ..We have discovered that the diffusible TGFss-related factor Radar (Gdf6a) is necessary and sufficient for activation of dorsal markers, such as Bmp4, Tbx5, Tbx2b, and Ephrin-B2, and ..
  2. Kruse Bend R, Rosenthal J, Quist T, Veien E, Fuhrmann S, Dorsky R, et al. Extraocular ectoderm triggers dorsal retinal fate during optic vesicle evagination in zebrafish. Dev Biol. 2012;371:57-65 pubmed publisher
    ..We identified gdf6a as a dorsal initiation signal acting from the extraocular non-neural ectoderm during optic vesicle evagination...
  3. Delot E, Kataoka H, Goutel C, Yan Y, Postlethwait J, Wittbrodt J, et al. The BMP-related protein radar: a maintenance factor for dorsal neuroectoderm cells?. Mech Dev. 1999;85:15-25 pubmed
    We have previously cloned several members of the TGF-beta superfamily of growth factors in zebrafish, one of which, Radar, belongs to the Dpp-Vg1-related (DVR) subgroup, with highest homology to GDF6...
  4. Goutel C, Kishimoto Y, Schulte Merker S, Rosa F. The ventralizing activity of Radar, a maternally expressed bone morphogenetic protein, reveals complex bone morphogenetic protein interactions controlling dorso-ventral patterning in zebrafish. Mech Dev. 2000;99:15-27 pubmed
    ..We provide evidence here that a maternally expressed bone morphogenetic protein (BMP), Radar, may control early ventral specification in zebrafish...
  5. den Hollander A, Biyanwila J, Kovach P, Bardakjian T, Traboulsi E, Ragge N, et al. Genetic defects of GDF6 in the zebrafish out of sight mutant and in human eye developmental anomalies. BMC Genet. 2010;11:102 pubmed publisher
    ..we show that the severe reduction of eye size in the outm233 mutant is caused by a mutation in the zebrafish gdf6a gene...
  6. Asai Coakwell M, French C, Ye M, Garcha K, Bigot K, Perera A, et al. Incomplete penetrance and phenotypic variability characterize Gdf6-attributable oculo-skeletal phenotypes. Hum Mol Genet. 2009;18:1110-21 pubmed publisher
    ..We identified seven heterozygous mutations in growth differentiation factor 6 (GDF6), a member of the BMP family, in patients with both ocular and vertebral anomalies, ..
  7. Nguyen Chi M, Bryson Richardson R, Sonntag C, Hall T, Gibson A, Sztal T, et al. Morphogenesis and cell fate determination within the adaxial cell equivalence group of the zebrafish myotome. PLoS Genet. 2012;8:e1003014 pubmed publisher
    ..Thus our results reveal that the synergistic actions of HH, FGF, and BMP signaling independently create a three-dimensional (3D) signaling milieu that coordinates cell fate within the adaxial cell equivalence group. ..
  8. French C, Erickson T, French D, Pilgrim D, Waskiewicz A. Gdf6a is required for the initiation of dorsal-ventral retinal patterning and lens development. Dev Biol. 2009;333:37-47 pubmed publisher
    ..In this study, we investigate the functions of two zebrafish Bmps, Gdf6a and Bmp4, during initiation of dorsal retinal identity, and subsequently during lens differentiation...
  9. 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. ..

More Information

Publications44

  1. Asai Coakwell M, March L, Dai X, Duval M, Lopez I, French C, et al. Contribution of growth differentiation factor 6-dependent cell survival to early-onset retinal dystrophies. Hum Mol Genet. 2013;22:1432-42 pubmed publisher
    ..Here we show that mutations in the transforming growth factor-? (TGF-?) ligand Growth Differentiation Factor 6, which specifies the dorso-ventral retinal axis, contribute to Leber congenital amaurosis...
  2. Crosier P, Kalev Zylinska M, Hall C, Flores M, Horsfield J, Crosier K. Pathways in blood and vessel development revealed through zebrafish genetics. Int J Dev Biol. 2002;46:493-502 pubmed
    ..Gdf6a/radar, a member of the bone morphogenetic protein (BMP) family, is expressed in the zebrafish hypochord and primitive ..
  3. Hall C, Flores M, Davidson A, Crosier K, Crosier P. Radar is required for the establishment of vascular integrity in the zebrafish. Dev Biol. 2002;251:105-17 pubmed
    ..Here, we propose a role for Radar, one of the zebrafish orthologues of gdf6, in establishing integrity of the trunk vasculature in zebrafish embryos...
  4. Sidi S, Goutel C, Peyrieras N, Rosa F. Maternal induction of ventral fate by zebrafish radar. Proc Natl Acad Sci U S A. 2003;100:3315-20 pubmed
    ..in the zebrafish embryo by the maternally encoded transforming growth factor beta (TGF-beta) signaling molecule, Radar. Loss-of-function experiments, together with epistasis analyses, identify maternal Radar as an upstream activator ..
  5. Asai Coakwell M, French C, Berry K, Ye M, Koss R, Somerville M, et al. GDF6, a novel locus for a spectrum of ocular developmental anomalies. Am J Hum Genet. 2007;80:306-15 pubmed
    ..deletion encompasses GDF6, encoding a member of the bone-morphogenetic protein family, and that inhibition of gdf6a in a model organism accurately recapitulates the proband's phenotype...
  6. French C, Erickson T, Callander D, Berry K, Koss R, Hagey D, et al. Pbx homeodomain proteins pattern both the zebrafish retina and tectum. BMC Dev Biol. 2007;7:85 pubmed
    ..Furthermore, we determined that some Pbx-dependent transcripts also require Meis1 and Gdf6a function...
  7. Latimer A, Appel B. Notch signaling regulates midline cell specification and proliferation in zebrafish. Dev Biol. 2006;298:392-402 pubmed
    ..Together, our results indicate that Notch signaling regulates allocation of appropriate numbers of different midline cells by different mechanisms. ..
  8. Valdivia L, Lamb D, Horner W, Wierzbicki C, Tafessu A, Williams A, et al. Antagonism between Gdf6a and retinoic acid pathways controls timing of retinal neurogenesis and growth of the eye in zebrafish. Development. 2016;143:1087-98 pubmed publisher
    ..These mutants carry lesions in gdf6a, a gene encoding a BMP family member previously implicated in dorsoventral patterning of the eye...
  9. Rissi M, Wittbrodt J, Delot E, Naegeli M, Rosa F. Zebrafish Radar: a new member of the TGF-beta superfamily defines dorsal regions of the neural plate and the embryonic retina. Mech Dev. 1995;49:223-34 pubmed
    ..new members of this superfamily involved in the control of early zebrafish embryogenesis, we have isolated a gene, Radar, which appears to be conserved throughout vertebrate evolution and defines a new subfamily within the superfamily...
  10. Wilm T, Solnica Krezel L. Radar breaks the fog: insights into dorsoventral patterning in zebrafish. Proc Natl Acad Sci U S A. 2003;100:4363-5 pubmed
  11. Ringholm A, Fredriksson R, Poliakova N, Yan Y, Postlethwait J, Larhammar D, et al. One melanocortin 4 and two melanocortin 5 receptors from zebrafish show remarkable conservation in structure and pharmacology. J Neurochem. 2002;82:6-18 pubmed
  12. Reed N, Mortlock D. Identification of a distant cis-regulatory element controlling pharyngeal arch-specific expression of zebrafish gdf6a/radar. Dev Dyn. 2010;239:1047-60 pubmed publisher
    ..b>radar/gdf6a is a zebrafish ortholog of Gdf6 and has an essential role in embryonic patterning...
  13. Qian L, Wang Y, Jiang Q, Zhong T, Song H. Ethanol disrupts the formation of hypochord and dorsal aorta during the development of embryonic zebrafish. Sci China C Life Sci. 2005;48:608-15 pubmed
    ..The consequent lack of vasculogenic factors including Radar and Ang-1 partly explains the defects in formation and integrity of dorsal aorta...
  14. Schebesta M, Lien C, Engel F, Keating M. Transcriptional profiling of caudal fin regeneration in zebrafish. ScientificWorldJournal. 2006;6 Suppl 1:38-54 pubmed
    ..Taken together, we provide a comprehensive database of fin regeneration that will serve as an important tool for understanding the molecular mechanisms of regeneration. ..
  15. Holly V, Widen S, Famulski J, Waskiewicz A. Sfrp1a and Sfrp5 function as positive regulators of Wnt and BMP signaling during early retinal development. Dev Biol. 2014;388:192-204 pubmed publisher
    ..Overexpression of a low dose of sfrp5 mRNA causes an increase in dorsal retina marker gene expression. We propose a model in which Sfrp proteins function as facilitators of both BMP and Wnt signaling within the dorsal retina. ..
  16. Hogan B, Layton J, Pyati U, Nutt S, Hayman J, Varma S, et al. Specification of the primitive myeloid precursor pool requires signaling through Alk8 in zebrafish. Curr Biol. 2006;16:506-11 pubmed
    ..1 expression, but the role of alk8 was independent of the scl-dependent cell-fate pathway. Furthermore, the role of Alk8 in myelopoiesis was genetically separable from its earlier role in dorsal-ventral embryonic patterning. ..
  17. Pant S, March L, Famulski J, French C, Lehmann O, Waskiewicz A. Molecular mechanisms regulating ocular apoptosis in zebrafish gdf6a mutants. Invest Ophthalmol Vis Sci. 2013;54:5871-9 pubmed publisher
    ..The role of Gdf6 in regulating apoptosis was studied using a zebrafish gdf6a(-/-) mutant, which encodes a truncated, nonfunctional protein...
  18. Maurus D, Harris W. Zic-associated holoprosencephaly: zebrafish Zic1 controls midline formation and forebrain patterning by regulating Nodal, Hedgehog, and retinoic acid signaling. Genes Dev. 2009;23:1461-73 pubmed publisher
    ..Repression of BMP signaling in dorsal forebrain has been implicated in causing MIH HPE. This work provides a mechanistical explanation at the molecular level of why Zic factors are associated with both major forms of HPE. ..
  19. David N, Saint Etienne L, Tsang M, Schilling T, Rosa F. Requirement for endoderm and FGF3 in ventral head skeleton formation. Development. 2002;129:4457-68 pubmed
    ..Together, our results reveal for the first time that the endoderm provides differential cues along the anteroposterior axis to control ventral head skeleton development and demonstrate that this function is mediated in part by Fgf3. ..
  20. Nojima H, Rothhämel S, Shimizu T, Kim C, Yonemura S, Marlow F, et al. Syntabulin, a motor protein linker, controls dorsal determination. Development. 2010;137:923-33 pubmed publisher
    ..Our findings suggest that Syntabulin regulates the microtubule-dependent transport of the DDs, and provide evidence for the link between AV and dorsoventral axis formation. ..
  21. Tanaka H, Morimura R, Ohshima T. Dpysl2 (CRMP2) and Dpysl3 (CRMP4) phosphorylation by Cdk5 and DYRK2 is required for proper positioning of Rohon-Beard neurons and neural crest cells during neurulation in zebrafish. Dev Biol. 2012;370:223-36 pubmed publisher
    ..These results suggest that the phosphorylation of Dpysl2 and Dpysl3 by Cdk5 and DYRK2 is required for the proper positioning of RB neurons and NCCs during neurulation in zebrafish embryos. ..
  22. Bonner J, Gribble S, Veien E, Nikolaus O, Weidinger G, Dorsky R. Proliferation and patterning are mediated independently in the dorsal spinal cord downstream of canonical Wnt signaling. Dev Biol. 2008;313:398-407 pubmed
    ..Together, our work demonstrates that proliferation and patterning in the developing spinal cord are separable events that are regulated independently by Wnt signaling. ..
  23. Erickson T, French C, Waskiewicz A. Meis1 specifies positional information in the retina and tectum to organize the zebrafish visual system. Neural Dev. 2010;5:22 pubmed publisher
    ..By patterning both the retina and tectum, Meis1 plays an important role in establishing the retinotectal map and organizing the visual system. ..
  24. Kinna G, Kolle G, Carter A, Key B, Lieschke G, Perkins A, et al. Knockdown of zebrafish crim1 results in a bent tail phenotype with defects in somite and vascular development. Mech Dev. 2006;123:277-87 pubmed
    ..Hence, the primary role of zebrafish crim1 is likely to be the regulation of somitic and vascular development. ..
  25. Chen L, Dong C, Kong S, Zhang J, Li X, Xu P. Genome wide identification, phylogeny, and expression of bone morphogenetic protein genes in tetraploidized common carp (Cyprinus carpio). Gene. 2017;627:157-163 pubmed publisher
    ..The whole set of bmp gene family in common carp provides insight into gene fate of tetraploidized common carp genome post recent WGD. ..
  26. Zhang Y, Qin W, Lu X, Xu J, Huang H, Bai H, et al. Programmable base editing of zebrafish genome using a modified CRISPR-Cas9 system. Nat Commun. 2017;8:118 pubmed publisher
    ..The use of base editing enables precise genetic modifications in model animals. Here the authors show high efficient single-base editing in zebrafish using modified Cas9 and its VQR variant with an altered PAM specificity. ..
  27. Elsen G, Choi L, Millen K, Grinblat Y, Prince V. Zic1 and Zic4 regulate zebrafish roof plate specification and hindbrain ventricle morphogenesis. Dev Biol. 2008;314:376-92 pubmed publisher
    ..In summary, we conclude that Zic1 and Zic4 control zebrafish 4th ventricle morphogenesis by regulating multiple mechanisms including cell proliferation and fate specification in the dorsal hindbrain. ..
  28. Ramel M, Hill C. The ventral to dorsal BMP activity gradient in the early zebrafish embryo is determined by graded expression of BMP ligands. Dev Biol. 2013;378:170-82 pubmed publisher
  29. Cermenati S, Moleri S, Cimbro S, Corti P, Del Giacco L, Amodeo R, et al. Sox18 and Sox7 play redundant roles in vascular development. Blood. 2008;111:2657-66 pubmed
    ..Our data suggest that a defect in arteriovenous identity could be responsible for the formation of telangiectases in patients with HLT. ..
  30. Duval M, Oel A, Allison W. gdf6a is required for cone photoreceptor subtype differentiation and for the actions of tbx2b in determining rod versus cone photoreceptor fate. PLoS ONE. 2014;9:e92991 pubmed publisher
    ..using nocturnal murine models, we instead utilize cone-rich zebrafish and herein investigate relationships between gdf6a and tbx2b in cone photoreceptor development...
  31. Peyrieras N, Strahle U, Rosa F. Conversion of zebrafish blastomeres to an endodermal fate by TGF-beta-related signaling. Curr Biol. 1998;8:783-6 pubmed
  32. Postlethwait J, Yan Y, Gates M, Horne S, Amores A, Brownlie A, et al. Vertebrate genome evolution and the zebrafish gene map. Nat Genet. 1998;18:345-9 pubmed
    ..This zebrafish gene map will facilitate molecular identification of mutated zebrafish genes, which can suggest functions for human genes known only by sequence. ..
  33. 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. ..
  34. Reichert S, Randall R, Hill C. A BMP regulatory network controls ectodermal cell fate decisions at the neural plate border. Development. 2013;140:4435-44 pubmed publisher
    ..Taken together, our data define a BMP regulatory network that controls cell fate decisions at the neural plate border. ..
  35. Davidson A, Postlethwait J, Yan Y, Beier D, Van Doren C, Foernzler D, et al. Isolation of zebrafish gdf7 and comparative genetic mapping of genes belonging to the growth/differentiation factor 5, 6, 7 subgroup of the TGF-beta superfamily. Genome Res. 1999;9:121-9 pubmed
    ..In zebrafish, there are three genes that belong to the Gdf5, 6, 7 subgroup that have been named radar, dynamo, and contact. The genes radar and dynamo both encode proteins most similar to mouse GDF6...