Journal article
The intraflagellar transport protein, IFT88, is essential for vertebrate photoreceptor assembly and maintenance
The Journal of cell biology, Vol.157(1), pp.103-114
04/01/2002
Handle:
https://hdl.handle.net/2376/104434
PMCID: PMC2173265
PMID: 11916979
Abstract
Approximately 10% of the photoreceptor outer segment (OS) is turned over each day, requiring large amounts of lipid and protein to be moved from the inner segment to the OS. Defects in intraphotoreceptor transport can lead to retinal degeneration and blindness. The transport mechanisms are unknown, but because the OS is a modified cilium, intraflagellar transport (IFT) is a candidate mechanism. IFT involves movement of large protein complexes along ciliary microtubules and is required for assembly and maintenance of cilia. We show that IFT particle proteins are localized to photoreceptor connecting cilia. We further find that mice with a mutation in the IFT particle protein gene, Tg737/IFT88, have abnormal OS development and retinal degeneration. Thus, IFT is important for assembly and maintenance of the vertebrate OS.
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Details
- Title
- The intraflagellar transport protein, IFT88, is essential for vertebrate photoreceptor assembly and maintenance
- Creators
- Gregory J Pazour - Department of Cell Biology, University of Massachusetts Medical School, Worcester, MA 01655Sheila A Baker - Department of Cell Biology, Neurobiology, and Anatomy, Medical College of Wisconsin, Milwaukee, WI 53226James A Deane - Department of Molecular, Cellular, and Developmental Biology, Yale University, New Haven, CT 06520Douglas G Cole - Department of Microbiology, Molecular Biology, and Biochemistry, University of Idaho, Moscow, ID 83844Bethany L Dickert - Department of Cell Biology, University of Massachusetts Medical School, Worcester, MA 01655Joel L Rosenbaum - Department of Molecular, Cellular, and Developmental Biology, Yale University, New Haven, CT 06520George B Witman - Department of Cell Biology, University of Massachusetts Medical School, Worcester, MA 01655Joseph C Besharse - Department of Cell Biology, Neurobiology, and Anatomy, Medical College of Wisconsin, Milwaukee, WI 53226
- Publication Details
- The Journal of cell biology, Vol.157(1), pp.103-114
- Academic Unit
- Center for Reproductive Biology
- Identifiers
- 99900546892001842
- Language
- English
- Resource Type
- Journal article