PUBLICATION
Genetic Deletion of Zebrafish Rab28 Causes Defective Outer Segment Shedding, but Not Retinal Degeneration
- Authors
- Carter, S.P., Moran, A.L., Matallanas, D., McManus, G.J., Blacque, O.E., Kennedy, B.N.
- ID
- ZDB-PUB-200422-52
- Date
- 2020
- Source
- Frontiers in cell and developmental biology 8: 136 (Journal)
- Registered Authors
- Kennedy, Breandan N.
- Keywords
- cilia, outer segment, photoreceptor, retinal degeneration, small G protein, visual function, zebrafish
- MeSH Terms
- none
- PubMed
- 32258030 Full text @ Front Cell Dev Biol
Abstract
The photoreceptor outer segment is the canonical example of a modified and highly specialized cilium, with an expanded membrane surface area in the form of disks or lamellae for efficient light detection. Many ciliary proteins are essential for normal photoreceptor function and cilium dysfunction often results in retinal degeneration leading to impaired vision. Herein, we investigate the function and localization of the ciliary G-protein RAB28 in zebrafish cone photoreceptors. CRISPR-Cas9 generated rab28 mutant zebrafish display significantly reduced shed outer segment material/phagosomes in the RPE at 1 month post fertilization (mpf), but otherwise normal visual function up to 21 dpf and retinal structure up to 12 mpf. Cone photoreceptor-specific transgenic reporter lines show Rab28 localizes almost exclusively to outer segments, independently of GTP/GDP nucleotide binding. Co-immunoprecipitation analysis demonstrates tagged Rab28 interacts with components of the phototransduction cascade, including opsins, phosphodiesterase 6C and guanylate cyclase 2D. Our data shed light on RAB28 function in cones and provide a model for RAB28-associated cone-rod dystrophy.
Genes / Markers
Figure Gallery (8 images)
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping