期刊
CELL BIOCHEMISTRY AND FUNCTION
卷 34, 期 6, 页码 429-440出版社
WILEY
DOI: 10.1002/cbf.3205
关键词
anterograde transport; Kif3a; outer segment; retinal degeneration; zebrafish
资金
- Royal Society of London
- National Eye Research Centre
- Visual Research Trust
- Fight for Sight
- Rosetrees Trust
- Yorkhill Children's Charity
- Saudi Arabia government PhD scholarship
- EU INTERREG NEW noPILLS program
- Biotechnology and Biological Sciences Research Council [BB/L00111X/1] Funding Source: researchfish
- Fight for Sight [URP12, 1419/20] Funding Source: researchfish
- Glasgow Children's Hospital Charity [YRSS/PSG/2014/06] Funding Source: researchfish
- Rosetrees Trust [M160-F1] Funding Source: researchfish
- BBSRC [BB/L00111X/1] Funding Source: UKRI
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.
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