Heterotrimeric Kinesin-2 (KIF3) Mediates Transition Zone and Axoneme Formation of Mouse Photoreceptors

Jiang, Li; Wei, Yuxiao; Ronquillo, Cecinio C.; Marc, Robert E.; Yoder, Bradley K.; Frederick, Jeanne M.; Baehr, Wolfgang

doi:10.1074/jbc.m115.638437

Cited by 58 publications

(83 citation statements)

References 63 publications

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“…Accordingly, mutations in the human ARL13b gene disabling GEF activity on ARL3 and germline knock-out of the mouse Arl13b gene cause Joubert syndrome (44,45). The ret Arl3 Ϫ/Ϫ phenotype is strikingly similar to a retinaspecific knock-out of KIF3A (obligatory subunit of the heterotrimeric kinesin-2, KIF3) and IFT88 (particle of the IFT-B complex) (60). KIF3 and IFT88 are responsible for IFT of tubulin and axoneme building blocks.…”

Section: Arl3mentioning

confidence: 89%

“…Six3-Cre-mediated recombination is known to be delayed in the retina periphery (60,62). We therefore tested CC/OS formation by rhodopsin immunohistochemistry in the presence of EGFP-CETN2 at P10 and P15, the times when ERG and OKT showed significant visual responses.…”

Section: Arl3mentioning

confidence: 99%

“…ret Kif3a Ϫ/Ϫ retina lacking connecting cilia and outer segments (60). Trafficking of lipidated proteins (PDE6) (Fig.…”

Section: Arl3mentioning

confidence: 99%

“…3D), a well characterized centriole (50) and connecting cilium marker (60). Transmembrane proteins, e.g.…”

Section: Arl3 Regulates Trafficking Of Lipidated Peripheral Proteins-mentioning

confidence: 99%

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Arf-like Protein 3 (ARL3) Regulates Protein Trafficking and Ciliogenesis in Mouse Photoreceptors

Hanke-Gogokhia

Gerstner

et al. 2016

Journal of Biological Chemistry

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112

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Section: Arl3mentioning

confidence: 89%

Section: Arl3mentioning

confidence: 99%

See 2 more Smart Citations

Arf-like Protein 3 (ARL3) Regulates Protein Trafficking and Ciliogenesis in Mouse Photoreceptors

Hanke-Gogokhia

Gerstner

et al. 2016

Journal of Biological Chemistry

Self Cite

112

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“…These results are consistent with a recent demonstration, that guanylate cycle-1 interacts with, and is co-transported and stabilized by rhodopsin (Pearring et al, 2015). The role of IFT in OS protein delivery has been questioned in studies by one group that had difficulty detecting significant defects in rhodopsin localization (Avasthi et al, 2009; Jiang et al, 2015). However, immunocytochemical studies of fixed tissue, showing end-state localization, have major drawbacks for studying dynamic cell biology, such as protein trafficking.…”

Section: Molecular Basis For Os Architecturementioning

confidence: 99%

Molecular basis for photoreceptor outer segment architecture

Goldberg

Moritz

Williams

2016

Progress in Retinal and Eye Research

162

160

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To serve vision, vertebrate rod and cone photoreceptors must detect photons, convert the light stimuli into cellular signals, and then convey the encoded information to downstream neurons. Rods and cones are sensory neurons that each rely on specialized ciliary organelles to detect light. These organelles, called outer segments, possess elaborate architectures that include many hundreds of light-sensitive membranous disks arrayed one atop another in precise register. These stacked disks capture light and initiate the chain of molecular and cellular events that underlie normal vision. Outer segment organization is challenged by an inherently dynamic nature; these organelles are subject to a renewal process that replaces a significant fraction of their disks (up to ~10%) on a daily basis. In addition, a broad range of environmental and genetic insults can disrupt outer segment morphology to impair photoreceptor function and viability. In this chapter, we survey the major progress that has been made for understanding the molecular basis of outer segment architecture. We also discuss key aspects of organelle lipid and protein composition, and highlight distributions, interactions, and potential structural functions of key OS-resident molecules, including: kinesin-2, actin, RP1, prominin-1, protocadherin 21, peripherin-2/rds, rom-1, glutamic acid-rich proteins, and rhodopsin. Finally, we identify key knowledge gaps and challenges that remain for understanding how normal outer segment architecture is established and maintained.

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

Raghupathy

Zhang

Alhasani

et al. 2016

Cell Biochemistry & Function

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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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Heterotrimeric Kinesin-2 (KIF3) Mediates Transition Zone and Axoneme Formation of Mouse Photoreceptors

Cited by 58 publications

References 63 publications

Arf-like Protein 3 (ARL3) Regulates Protein Trafficking and Ciliogenesis in Mouse Photoreceptors

Arf-like Protein 3 (ARL3) Regulates Protein Trafficking and Ciliogenesis in Mouse Photoreceptors

Molecular basis for photoreceptor outer segment architecture

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

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