PUBLICATION

Cilia-driven fluid flow in the zebrafish pronephros, brain and Kupffer's vesicle is required for normal organogenesis

Authors
Kramer-Zucker, A.G., Olale, F., Haycraft, C.J., Yoder, B.K., Schier, A.F., and Drummond, I.A.
ID
ZDB-PUB-050328-4
Date
2005
Source
Development (Cambridge, England)   132(8): 1907-1921 (Journal)
Registered Authors
Drummond, Iain, Kramer-Zucker, Albrecht, Olale, Felix, Schier, Alexander
Keywords
Cilia, Pronephros, Kupffer's vesicle, Ependymal cell, Spinal canal, Kidney cyst, Hydrocephalus, Left-right asymmetry
MeSH Terms
  • Microscopy, Electron
  • Nerve Tissue Proteins/genetics
  • Base Sequence
  • Immunohistochemistry
  • Cilia/physiology*
  • Dyneins/genetics
  • Organogenesis/physiology*
  • Animals
  • Molecular Sequence Data
  • Embryo, Nonmammalian/embryology
  • Embryo, Nonmammalian/ultrastructure
  • Zebrafish/embryology*
  • Organizers, Embryonic/embryology*
  • Oligonucleotides
  • Body Patterning/physiology
  • Central Nervous System/embryology*
  • Microscopy, Video
  • Sequence Analysis, DNA
  • Kidney/embryology*
  • DNA Primers
  • Microscopy, Fluorescence
  • In Situ Hybridization
  • Cloning, Molecular
  • Body Fluids/physiology*
(all 24)
PubMed
15790966 Full text @ Development
Abstract
Cilia, as motile and sensory organelles, have been implicated in normal development, as well as diseases including cystic kidney disease, hydrocephalus and situs inversus. In kidney epithelia, cilia are proposed to be non-motile sensory organelles, while in the mouse node, two cilia populations, motile and non-motile have been proposed to regulate situs. We show that cilia in the zebrafish larval kidney, the spinal cord and Kupffer's vesicle are motile, suggesting that fluid flow is a common feature of each of these organs. Disruption of cilia structure or motility resulted in pronephric cyst formation, hydrocephalus and left-right asymmetry defects. The data show that loss of fluid flow leads to fluid accumulation, which can account for organ distension pathologies in the kidney and brain. In Kupffer's vesicle, loss of flow is associated with loss of left-right patterning, indicating that the `nodal flow' mechanism of generating situs is conserved in non-mammalian vertebrates.
Genes / Markers
Figures
Figure Gallery (10 images)
Show all Figures
Expression
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
tz288
    		Point Mutation
    1 - 1 of 1
    Show
    Human Disease / Model
    No data available
    Sequence Targeting Reagents
    Target Reagent Reagent Type
    dnah9lMO1-dnah9lMRPHLNO
    ift57MO1-ift57MRPHLNO
    ift57MO2-ift57MRPHLNO
    ift88MO1-ift88MRPHLNO
    ift88MO2-ift88MRPHLNO
    1 - 5 of 5
    Show
    Fish
    Fish
    ift88tz288/tz288
    WT
    WT + MO1-ift88
    1 - 3 of 3
    Show
    Antibodies
    Orthology
    No data available
    Engineered Foreign Genes
    No data available
    Mapping
    No data available
    Your Input Welcome
    We welcome your input and comments. Please use this form to recommend updates to the information in ZFIN. We appreciate as much detail as possible and references as appropriate. We will review your comments promptly.
    Citation
    Kramer-Zucker, A.G., Olale, F., Haycraft, C.J., Yoder, B.K., Schier, A.F., and Drummond, I.A. (2005) Cilia-driven fluid flow in the zebrafish pronephros, brain and Kupffer's vesicle is required for normal organogenesis. Development (Cambridge, England). 132(8):1907-1921.