PUBLICATION
Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish
- Authors
- Raghupathy, R.K., Zhang, X., Alhasani, R.H., Zhou, X., Mullin, M., Reilly, J., Li, W., Liu, M., Shu, X.
- ID
- ZDB-PUB-160730-7
- Date
- 2016
- Source
- Cell biochemistry and function 34(6): 429-40 (Journal)
- Registered Authors
- Liu, Mugen
- Keywords
- Kif3a, anterograde transport, outer segment, retinal degeneration, zebrafish
- MeSH Terms
-
- Animals
- Base Sequence
- Caffeine/pharmacology
- Cryoultramicrotomy
- Gene Expression Regulation, Developmental/drug effects
- In Situ Nick-End Labeling
- Intracellular Space/metabolism
- Kinesins/genetics*
- Kinesins/metabolism
- Melanosomes/drug effects
- Melanosomes/metabolism
- Mutation/genetics*
- Phenotype
- Protein Transport/drug effects
- Retinal Degeneration/metabolism*
- Retinal Degeneration/pathology*
- Retinal Photoreceptor Cell Outer Segment/metabolism*
- Retinal Photoreceptor Cell Outer Segment/pathology*
- Zebrafish/embryology
- Zebrafish/genetics*
- Zebrafish Proteins/genetics*
- Zebrafish Proteins/metabolism
- PubMed
- 27470972 Full text @ Cell Biochem. Funct.
Citation
Raghupathy, R.K., Zhang, X., Alhasani, R.H., Zhou, X., Mullin, M., Reilly, J., Li, W., Liu, M., Shu, X. (2016) Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish. Cell biochemistry and function. 34(6):429-40.
Abstract
Photoreceptors are highly specialized sensory neurons that possess a modified primary cilium called the outer segment. Photoreceptor outer segment formation and maintenance require highly active protein transport via a process known as intraflagellar transport. Anterograde transport in outer segments is powered by the heterotrimeric kinesin II and coordinated by intraflagellar transport proteins. Here, we describe a new zebrafish model carrying a nonsense mutation in the kinesin II family member 3A (kif3a) gene. Kif3a mutant zebrafish exhibited curved body axes and kidney cysts. Outer segments were not formed in most parts of the mutant retina, and rhodopsin was mislocalized, suggesting KIF3A has a role in rhodopsin trafficking. Both rod and cone photoreceptors degenerated rapidly between 4 and 9 days post fertilization, and electroretinography response was not detected in 7 days post fertilization mutant larvae. Loss of KIF3A in zebrafish also resulted in an intracellular transport defect affecting anterograde but not retrograde transport of organelles. Our results indicate KIF3A plays a conserved role in photoreceptor outer segment formation and intracellular transport.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping