Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish. (29th July 2016)

• Record Type: Journal Article
• Authors: Raghupathy, Rakesh K.; Zhang, Xun; Alhasani, Reem H.; Zhou, Xinzhi; Mullin, Margaret; Reilly, James; Li, Wenchang; Liu, Mugen; Shu, Xinhua
• Is Part Of: Cell biochemistry and function. Volume 34:Number 6(2016)
• Page Start: 429
• Page End: 440
• Publication Date: 2016-07-29
• Subjects: anterograde transport -- Kif3a -- outer segment -- retinal degeneration -- zebrafish; Cytochemistry -- Periodicals; Cell metabolism -- Periodicals; Biochemistry -- Periodicals; Cytology -- Periodicals; 572
• DOI: 10.1002/cbf.3205
• Languages: English
• ISSNs: 0263-6484

Abstract

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.