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
- Carrier Proteins/genetics*
- Phalloidine
- Mice
- Molecular Sequence Data
- Embryo, Nonmammalian
- Retinal Rod Photoreceptor Cells/metabolism
- Chromosome Mapping
- Retina/metabolism
- Retina/ultrastructure
- Animals, Genetically Modified
- Animals
- Microinjections
- Phenotype
- Embryo, Mammalian
- RNA, Messenger/biosynthesis
- Sequence Homology, Amino Acid
- Blotting, Western/methods
- Plant Proteins
- Algal Proteins/physiology
- Cloning, Molecular/methods
- Cell Differentiation/genetics
- Cell Differentiation/physiology*
- Cilia/metabolism*
- Flagella/metabolism
- Neurons, Afferent/cytology*
- Neurons, Afferent/ultrastructure
- Luminescent Proteins/metabolism
- Retinal Cone Photoreceptor Cells/metabolism
- Cell Survival/genetics
- Cell Survival/physiology*
- Tubulin/metabolism
- In Situ Nick-End Labeling/methods
- Photic Stimulation
- Zebrafish
- Gene Expression Regulation, Developmental
- Biological Transport/genetics
- Biological Transport/physiology
- Green Fluorescent Proteins
- Body Patterning/genetics
- Body Patterning/physiology
- Propidium
- In Situ Hybridization/methods
- Protozoan Proteins/genetics
- Protozoan Proteins/physiology*
- Humans
- Mutation
- Microscopy, Electron
- Reverse Transcriptase Polymerase Chain Reaction/methods
- Immunohistochemistry/methods
- Sequence Analysis, DNA
- PubMed
- 15182712 Full text @ Neuron
Citation
Tsujikawa, M., and Malicki, J. (2004) Intraflagellar transport genes are essential for differentiation and survival of vertebrate sensory neurons. Neuron. 42(5):703-716.
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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping