September 2016
Volume 57, Issue 12
Open Access
ARVO Annual Meeting Abstract  |   September 2016
Primary Cilia Temporally Regulate RPE Maturation via Regulation of WNT Signaling
Author Affiliations & Notes
  • Helen May-Simera
    Zoology, Johannes Gutenberg University, Mainz, Germany
  • Sarita Patnaik
    Zoology, Johannes Gutenberg University, Mainz, Germany
  • Viola Kretschmer
    Zoology, Johannes Gutenberg University, Mainz, Germany
  • Kapil Bharti
    National Eye Institute, Unit on Ocular and Stem Cell Translational Research, Bethesda, Maryland, United States
  • Footnotes
    Commercial Relationships   Helen May-Simera, None; Sarita Patnaik, None; Viola Kretschmer, None; Kapil Bharti, None
  • Footnotes
    Support  Alexander Von Humboldt Foundation
Investigative Ophthalmology & Visual Science September 2016, Vol.57, 5416. doi:
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      Helen May-Simera, Sarita Patnaik, Viola Kretschmer, Kapil Bharti; Primary Cilia Temporally Regulate RPE Maturation via Regulation of WNT Signaling. Invest. Ophthalmol. Vis. Sci. 2016;57(12):5416.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose : The retinal pigment epithelium (RPE), a monolayer epithelium tightly associated with photoreceptors, is critical for photoreceptor function and survival. Development and function of the RPE has been shown to depend on wingless (Wnt) signaling, a core signaling pathway known to regulate diverse processes during development. Virtually all eukaryotic cells have a primary cilium, and in recent years the importance of this cellular organelle has reemerged with the discovery of its involvement in human developmental and degenerative disorders. One of the most important functions of primary cilia is to regulate signaling pathways.

Methods : Using a variety of microscopic and expression analyses, we examined ciliogenesis in the developing RPE in control and cilia mutant mice.

Results : We show that primary cilia are transiently expressed in the developing RPE and that they are required for the regulation of Wnt signaling. Primary cilia mutant mice have immature RPE and continually active canonical-WNT signaling in post-natal animals.

Conclusions : Our results show a developmental role for primary cilium in RPE maturation and provide insight into retinal degeneration caused by ciliopathies.

This is an abstract that was submitted for the 2016 ARVO Annual Meeting, held in Seattle, Wash., May 1-5, 2016.

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