Early defects in photoreceptor outer segment morphogenesis in zebrafish ift57, ift88 and ift172 Intraflagellar Transport mutants

Intraflagellar Transport (IFT) refers to a highly conserved process occurring in eukaryotic ciliated structures. In vertebrate photoreceptors, IFT mediates protein trafficking to the outer segments. The IFT particle is a multi-subunit complex and mutations in many individual components causes photor...

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Bibliographic Details
Published in:Vision research (Oxford) 2009-02, Vol.49 (4), p.479-489
Main Authors: Sukumaran, Sujita, Perkins, Brian D.
Format: Article
Language:English
Subjects:
Adaptor Proteins, Signal Transducing - genetics
Animals
Biological and medical sciences
Connecting cilium
Danio rerio
Embryo, Nonmammalian - anatomy & histology
Eye and associated structures. Visual pathways and centers. Vision
Fundamental and applied biological sciences. Psychology
Immunohistochemistry
Intraflagellar transport
Microscopy, Electron, Transmission
Morphogenesis - genetics
Mutation
Outer segment
Photoreceptor
Retina
Retinal Photoreceptor Cell Outer Segment - ultrastructure
Time Factors
Vertebrates: nervous system and sense organs
Zebrafish - embryology
Zebrafish - genetics
Zebrafish Proteins - genetics
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Summary:Intraflagellar Transport (IFT) refers to a highly conserved process occurring in eukaryotic ciliated structures. In vertebrate photoreceptors, IFT mediates protein trafficking to the outer segments. The IFT particle is a multi-subunit complex and mutations in many individual components causes photoreceptor defects. In zebrafish, mutations in the ift57, ift88, and ift172 genes result in retinal degeneration by 5 days post fertilization (dpf). Although the effects of these mutations on photoreceptor survival have been described, early developmental morphogenesis remains poorly understood. We used transmission electron microscopy and immunohistochemistry to examine these mutants at 60, 72, and 96 h post fertilization (hpf) and describe early photoreceptor morphogenesis defects.
ISSN:0042-6989
1878-5646
DOI:10.1016/j.visres.2008.12.009