PUBLICATION
Mutations in the Dynein1 Complex are Permissible for Basal Body Migration in Photoreceptors but Alter Rab6 Localization
- Authors
- Fogerty, J., Denton, K., Perkins, B.D.
- ID
- ZDB-PUB-151003-11
- Date
- 2016
- Source
- Advances in experimental medicine and biology 854: 209-15 (Chapter)
- Registered Authors
- Fogerty, Joseph, Perkins, Brian
- Keywords
- Basal body, Dynein, Rab, Retinal development, Zebrafish
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Basal Bodies/metabolism*
- Basal Bodies/ultrastructure
- Cilia/genetics
- Cilia/metabolism
- Cytoplasmic Dyneins/genetics*
- Cytoplasmic Dyneins/metabolism
- Dynactin Complex
- Embryo, Nonmammalian/embryology
- Embryo, Nonmammalian/metabolism
- Golgi Apparatus/metabolism
- Larva/genetics
- Larva/metabolism
- Luminescent Proteins/genetics
- Luminescent Proteins/metabolism
- Microscopy, Electron, Transmission
- Microscopy, Fluorescence
- Microtubule-Associated Proteins/genetics
- Microtubule-Associated Proteins/metabolism
- Mutation*
- Photoreceptor Cells, Vertebrate/metabolism*
- Protein Transport
- Zebrafish/embryology
- Zebrafish/genetics
- Zebrafish/metabolism
- Zebrafish Proteins/genetics*
- Zebrafish Proteins/metabolism
- rab GTP-Binding Proteins/metabolism*
- PubMed
- 26427413 Full text @ Adv. Exp. Med. Biol.
Citation
Fogerty, J., Denton, K., Perkins, B.D. (2016) Mutations in the Dynein1 Complex are Permissible for Basal Body Migration in Photoreceptors but Alter Rab6 Localization. Advances in experimental medicine and biology. 854:209-15.
Abstract
The photoreceptor outer segment is a specialized primary cilium, and anchoring of the basal body at the apical membrane is required for outer segment formation. We hypothesized that basal body localization and outer segment formation would require the microtubule motor dynein 1 and analyzed the zebrafish cannonball and mike oko mutants, which carry mutations in the heavy chain subunit of cytoplasmic dynein 1 (dync1h1) and the p150(Glued) subunit of Dynactin (dctn1a). The distribution of Rab6, a player in the post-Golgi trafficking of rhodopsin, was also examined. Basal body docking was unaffected in both mutants, but Rab6 expression was reduced. The results suggest that dynein 1 is dispensable for basal body docking but that outer segment defects may be due to defects in post-Golgi trafficking.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping