PUBLICATION

Intraflagellar transport genes are essential for differentiation and survival of vertebrate sensory neurons

Authors
Tsujikawa, M., and Malicki, J.
ID
ZDB-PUB-040609-12
Date
2004
Source
Neuron   42(5): 703-716 (Journal)
Registered Authors
Malicki, Jarema, Tsujikawa, Motokazu
Keywords
none
MeSH Terms
  • Rod Opsins/metabolism
  • Embryo, Mammalian
  • Phenotype
  • Photic Stimulation
  • Sequence Analysis, DNA
  • Cell Survival/genetics
  • Cell Survival/physiology*
  • Propidium
  • Mice
  • Flagella/metabolism
  • Reverse Transcriptase Polymerase Chain Reaction/methods
  • Sequence Homology, Amino Acid
  • Green Fluorescent Proteins
  • Phalloidine
  • Mutation
  • Cell Differentiation/genetics
  • Cell Differentiation/physiology*
  • Plant Proteins
  • Gene Expression Regulation, Developmental
  • Animals
  • Biological Transport/genetics
  • Biological Transport/physiology
  • Luminescent Proteins/metabolism
  • In Situ Hybridization/methods
  • Chromosome Mapping
  • Microinjections
  • Blotting, Western/methods
  • Retinal Rod Photoreceptor Cells/metabolism
  • Body Patterning/genetics
  • Body Patterning/physiology
  • Immunohistochemistry/methods
  • Humans
  • Retinal Cone Photoreceptor Cells/metabolism
  • Embryo, Nonmammalian
  • In Situ Nick-End Labeling/methods
  • Animals, Genetically Modified
  • RNA, Messenger/biosynthesis
  • Cilia/metabolism*
  • Zebrafish
  • Retina/metabolism
  • Retina/ultrastructure
  • Algal Proteins/physiology
  • Tubulin/metabolism
  • Cloning, Molecular/methods
  • Microscopy, Electron
  • Molecular Sequence Data
  • Carrier Proteins/genetics*
  • Protozoan Proteins/genetics
  • Protozoan Proteins/physiology*
  • Neurons, Afferent/cytology*
  • Neurons, Afferent/ultrastructure
PubMed
15182712 Full text @ Neuron
Abstract
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.
Genes / Markers
Figures
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping