ZFIN ID: ZDB-PUB-040609-12
Intraflagellar transport genes are essential for differentiation and survival of vertebrate sensory neurons
Tsujikawa, M., and Malicki, J.
Date: 2004
Source: Neuron   42(5): 703-716 (Journal)
Registered Authors: Malicki, Jarema, Tsujikawa, Motokazu
Keywords: none
MeSH Terms:
  • Algal Proteins/physiology
  • Animals
  • Animals, Genetically Modified
  • Biological Transport/genetics
  • Biological Transport/physiology
  • Blotting, Western/methods
  • Body Patterning/genetics
  • Body Patterning/physiology
  • Carrier Proteins/genetics*
  • Cell Differentiation/genetics
  • Cell Differentiation/physiology*
  • Cell Survival/genetics
  • Cell Survival/physiology*
  • Chromosome Mapping
  • Cilia/metabolism*
  • Cloning, Molecular/methods
  • Embryo, Mammalian
  • Embryo, Nonmammalian
  • Flagella/metabolism
  • Gene Expression Regulation, Developmental
  • Green Fluorescent Proteins
  • Humans
  • Immunohistochemistry/methods
  • In Situ Hybridization/methods
  • In Situ Nick-End Labeling/methods
  • Luminescent Proteins/metabolism
  • Mice
  • Microinjections
  • Microscopy, Electron
  • Molecular Sequence Data
  • Mutation
  • Neurons, Afferent/cytology*
  • Neurons, Afferent/ultrastructure
  • Phalloidine
  • Phenotype
  • Photic Stimulation
  • Plant Proteins
  • Propidium
  • Protozoan Proteins/genetics
  • Protozoan Proteins/physiology*
  • RNA, Messenger/biosynthesis
  • Retina/metabolism
  • Retina/ultrastructure
  • Retinal Cone Photoreceptor Cells/metabolism
  • Retinal Rod Photoreceptor Cells/metabolism
  • Reverse Transcriptase Polymerase Chain Reaction/methods
  • Rod Opsins/metabolism
  • Sequence Analysis, DNA
  • Sequence Homology, Amino Acid
  • Tubulin/metabolism
  • Zebrafish
PubMed: 15182712 Full text @ Neuron
Cilia play diverse roles in vertebrate and invertebrate sensory neurons. We show that a mutation of the zebrafish oval (ovl) locus affects a component of the ciliary transport (IFT) mechanism, the IFT88 polypeptide. In mutant retina, cilia are generated but not maintained, producing the absence of photoreceptor outer segments. A loss of cilia also occurs in auditory hair cells and olfactory sensory neurons. In all three sense organs, cilia defects are followed by degeneration of sensory cells. Similar phenotypes are induced by the absence of the IFT complex B polypeptides, ift52 and ift57, but not by the loss of complex A protein, ift140. The degeneration of mutant photoreceptor cells is caused, at least partially, by the ectopic accumulation of opsins. These studies reveal an essential role for IFT genes in vertebrate sensory neurons and implicate the molecular components of intraflagellar transport in degenerative disorders of these cells.