PUBLICATION

Lethal giant larvae 2 regulates development of the ciliated organ Kupffer's vesicle

Authors
Tay, H.G., Schulze, S.K., Compagnon, J., Foley, F.C., Heisenberg, C.P., Yost, H.J., Abdelilah-Seyfried, S., and Amack, J.D.
ID
ZDB-PUB-130403-12
Date
2013
Source
Development (Cambridge, England)   140(7): 1550-1559 (Journal)
Registered Authors
Abdelilah-Seyfried, Salim, Amack, Jeffrey, Compagnon, Julien, Foley, Fiona, Heisenberg, Carl-Philipp, Tay, Hwee Goon, Yost, H. Joseph
Keywords
cilia, lumenogenesis, lethal giant larvae 2, Rab11a, left-right asymmetry, zebrafish
MeSH Terms
  • Body Patterning/genetics
  • Morphogenesis/genetics*
  • Morphogenesis/physiology
  • Embryo, Nonmammalian
  • Embryonic Development/genetics
  • Embryonic Development/physiology
  • Animals, Genetically Modified
  • Animals
  • Larva/genetics
  • Larva/metabolism
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
  • Zebrafish Proteins/physiology*
  • Cell Polarity/genetics
  • Zebrafish*/embryology
  • Zebrafish*/genetics
  • Cilia/genetics
  • Cilia/metabolism
  • Cilia/physiology*
  • Gene Expression Regulation, Developmental
  • Kupffer Cells/metabolism
  • Kupffer Cells/physiology*
(all 22)
PubMed
23482490 Full text @ Development
Abstract

Motile cilia perform crucial functions during embryonic development and throughout adult life. Development of organs containing motile cilia involves regulation of cilia formation (ciliogenesis) and formation of a luminal space (lumenogenesis) in which cilia generate fluid flows. Control of ciliogenesis and lumenogenesis is not yet fully understood, and it remains unclear whether these processes are coupled. In the zebrafish embryo, lethal giant larvae 2 (lgl2) is expressed prominently in ciliated organs. Lgl proteins are involved in establishing cell polarity and have been implicated in vesicle trafficking. Here, we identified a role for Lgl2 in development of ciliated epithelia in Kupffer?s vesicle, which directs left-right asymmetry of the embryo; the otic vesicles, which give rise to the inner ear; and the pronephric ducts of the kidney. Using Kupffer?s vesicle as a model ciliated organ, we found that depletion of Lgl2 disrupted lumen formation and reduced cilia number and length. Immunofluorescence and time-lapse imaging of Kupffer?s vesicle morphogenesis in Lgl2-deficient embryos suggested cell adhesion defects and revealed loss of the adherens junction component E-cadherin at lateral membranes. Genetic interaction experiments indicate that Lgl2 interacts with Rab11a to regulate E-cadherin and mediate lumen formation that is uncoupled from cilia formation. These results uncover new roles and interactions for Lgl2 that are crucial for both lumenogenesis and ciliogenesis and indicate that these processes are genetically separable in zebrafish.

Genes / Markers
Figures
Figure Gallery (12 images) / 2
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
e102TgTransgenic Insertion
    mb1TgTransgenic Insertion
      pt19TgTransgenic Insertion
        s870TgTransgenic Insertion
          twu34TgTransgenic Insertion
            1 - 5 of 5
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            Human Disease / Model
            No data available
            Sequence Targeting Reagents
            Target Reagent Reagent Type
            llgl2MO1-llgl2MRPHLNO
            llgl2MO3-llgl2MRPHLNO
            rab11aMO1-rab11aMRPHLNO
            tp53MO4-tp53MRPHLNO
            1 - 4 of 4
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            Fish
            Antibodies
            Orthology
            No data available
            Engineered Foreign Genes
            Marker Marker Type Name
            EGFPEFGEGFP
            GFPEFGGFP
            TomatoEFGTomato
            1 - 3 of 3
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            Mapping
            No data available