PUBLICATION

Dampened Hedgehog signaling but normal Wnt signaling in zebrafish without cilia

Authors
Huang, P., and Schier, A.F.
ID
ZDB-PUB-090828-14
Date
2009
Source
Development (Cambridge, England)   136(18): 3089-3098 (Journal)
Registered Authors
Huang, Peng, Schier, Alexander
Keywords
Cilia, Hedgehog signaling, Wnt signaling, Gli, Spinal cord, Somite, Zebrafish
MeSH Terms
  • Hedgehog Proteins/genetics
  • Hedgehog Proteins/metabolism*
  • Cilia/metabolism*
  • Epistasis, Genetic
  • Oligonucleotides, Antisense
  • Oncogene Proteins/genetics
  • Oncogene Proteins/metabolism
  • Body Patterning
  • Animals, Genetically Modified
  • Trans-Activators/genetics
  • Trans-Activators/metabolism
  • Animals
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
  • Signal Transduction/physiology*
  • Zebrafish*/anatomy & histology
  • Zebrafish*/embryology
  • Zebrafish*/metabolism
  • In Situ Hybridization
  • Wnt Proteins/genetics
  • Wnt Proteins/metabolism*
(all 21)
PubMed
19700616 Full text @ Development
Abstract
Cilia have been implicated in Hedgehog (Hh) and Wnt signaling in mouse but not in Drosophila. To determine whether the role of cilia is conserved in zebrafish, we generated maternal-zygotic (MZ) oval (ovl; ift88) mutants that lack all cilia. MZovl mutants display normal canonical and non-canonical Wnt signaling but show defects in Hh signaling. As in mouse, zebrafish cilia are required to mediate the activities of Hh, Ptc, Smo and PKA. However, in contrast to mouse Ift88 mutants, which show a dramatic reduction in Hh signaling, zebrafish MZovl mutants display dampened, but expanded, Hh pathway activity. This activity is largely due to gli1, the expression of which is fully dependent on Hh signaling in mouse but not in zebrafish. These results reveal a conserved requirement for cilia in transducing the activity of upstream regulators of Hh signaling but distinct phenotypic effects due to differential regulation and differing roles of transcriptional mediators.
Genes / Markers
Figures
Figure Gallery (13 images) / 2
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Expression
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
a4594TgTransgenic Insertion
    ts294e
      		Point Mutation
      tz288
        		Point Mutation
        1 - 3 of 3
        Show
        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        Target Reagent Reagent Type
        dnd1MO1-dnd1MRPHLNO
        gli1MO1-gli1MRPHLNO
        gli2aMO1-gli2aMRPHLNO
        gli2bMO1-gli2bMRPHLNO
        gli3MO1-gli3MRPHLNO
        ptch1MO1-ptch1MRPHLNO
        ptch2MO3-ptch2MRPHLNO
        tp53MO4-tp53MRPHLNO
        1 - 8 of 8
        Show
        Fish
        Fish
        a4594Tg
        dzip1ts294e/ts294e
        ift88tz288/tz288
        1 - 3 of 3
        Show
        Antibodies
        Orthology
        No data available
        Engineered Foreign Genes
        Marker Marker Type Name
        GFPEFGGFP
        1 - 1 of 1
        Show
        Mapping
        No data available
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        Citation
        Huang, P., and Schier, A.F. (2009) Dampened Hedgehog signaling but normal Wnt signaling in zebrafish without cilia. Development (Cambridge, England). 136(18):3089-3098.