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

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 p...

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Vydané v:	Neuron (Cambridge, Mass.) Ročník 42; číslo 5; s. 703
Hlavní autori:	Tsujikawa, Motokazu, Malicki, Jarema
Médium:	Journal Article
Jazyk:	English
Vydavateľské údaje:	United States 10.06.2004
Predmet:	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 Retina - metabolism Retina - ultrastructure Retinal Cone Photoreceptor Cells - metabolism Retinal Rod Photoreceptor Cells - metabolism Reverse Transcriptase Polymerase Chain Reaction - methods RNA, Messenger - biosynthesis Rod Opsins - metabolism Sequence Analysis, DNA Sequence Homology, Amino Acid Tubulin - metabolism Zebrafish
ISSN:	0896-6273
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Shrnutí:	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.
Bibliografia:	ObjectType-Article-2 SourceType-Scholarly Journals-1 ObjectType-Feature-1 content type line 23
ISSN:	0896-6273
DOI:	10.1016/S0896-6273(04)00268-5