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ZFIN ID: ZDB-PUB-160730-7
Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish
Raghupathy, R.K., Zhang, X., Alhasani, R.H., Zhou, X., Mullin, M., Reilly, J., Li, W., Liu, M., Shu, X.
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
  • Kinesin/genetics*
  • Kinesin/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.
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.