Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish

Rakesh K. Raghupathy, Xun Zhang, Reem H. Alhasani, Xinzhi Zhou, Margaret Mullen, James Reilly, Wenchang Li, Mugen Liu, Xinhua Shu

Research output: Contribution to journalArticle

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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.
Original languageEnglish
Pages (from-to)429–440
Number of pages12
JournalCell Biochemistry and Function
Volume34
Issue number6
Early online date29 Jul 2016
DOIs
Publication statusPublished - Aug 2016

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Retinal Degeneration
Rhodopsin
Zebrafish
Fertilization
Neurons
Retinal Cone Photoreceptor Cells
Retinal Rod Photoreceptor Cells
Electroretinography
Cones
Carrier Proteins
Vertebrate Photoreceptor Cells
Cilia
Genes
Nonsense Codon
Active Biological Transport
Sensory Receptor Cells
Protein Transport
Organelles
Defects
Larva

Keywords

  • retinal regeneration
  • photoreceptor
  • zebrafish model
  • zebrafish

Cite this

Raghupathy, Rakesh K. ; Zhang, Xun ; Alhasani, Reem H. ; Zhou, Xinzhi ; Mullen, Margaret ; Reilly, James ; Li, Wenchang ; Liu, Mugen ; Shu, Xinhua. / Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish. In: Cell Biochemistry and Function. 2016 ; Vol. 34, No. 6. pp. 429–440.
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Abnormal photoreceptor outer segment development and early retinal degeneration in kif3a mutant zebrafish. / Raghupathy, Rakesh K.; Zhang, Xun; Alhasani, Reem H.; Zhou, Xinzhi; Mullen, Margaret; Reilly, James; Li, Wenchang; Liu, Mugen; Shu, Xinhua.

In: Cell Biochemistry and Function, Vol. 34, No. 6, 08.2016, p. 429–440.

Research output: Contribution to journalArticle

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AU - Reilly, James

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