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Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish
Author(s) -
Raghupathy Rakesh K.,
Zhang Xun,
Alhasani Reem H.,
Zhou Xinzhi,
Mullin Margaret,
Reilly James,
Li Wenchang,
Liu Mugen,
Shu Xinhua
Publication year - 2016
Publication title -
cell biochemistry and function
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.933
H-Index - 61
eISSN - 1099-0844
pISSN - 0263-6484
DOI - 10.1002/cbf.3205
Subject(s) - intraflagellar transport , zebrafish , cilium , microbiology and biotechnology , biology , kinesin , opsin , ciliogenesis , mutant , retinal degeneration , rhodopsin , retina , retinal , microtubule , genetics , neuroscience , biochemistry , gene
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.