Primary cilia dynamics instruct tissue patterning and repair of corneal endothelium

Andrea L. Blitzer, Lampros Panagis, G. Luca Gusella, John Danias, Marek Mlodzik, Carlo Iomini

Research output: Contribution to journalArticlepeer-review

37 Scopus citations


Primary cilia are required for several signaling pathways, but their function in cellular morphogenesis is poorly understood. Here we show that emergence of an hexagonal cellular pattern during development of the corneal endothelium (CE), a monolayer of neural crest-derived cells that maintains corneal transparency, depends on a precise temporal control of assembly of primary cilia that subsequently disassemble in adult corneal endothelial cells (CECs). However, cilia reassembly occurs rapidly in response to an in vivo mechanical injury and precedes basal body polarization and cellular elongation in mature CECs neighboring the wound. In contrast, CE from hypomorphic IFT88 mutants (Tg737orpk) or following in vivo lentiviral-mediated IFT88 knockdown display dysfunctional cilia and show disorganized patterning, mislocalization of junctional markers, and accumulation of cytoplasmic acetylated tubulin. Our results indicate an active role of cilia in orchestrating coordinated morphogenesis of CECs during development and repair and define the murine CE as a powerful in vivo system to study ciliary-based cellular dynamics.

Original languageEnglish (US)
Pages (from-to)2819-2824
Number of pages6
JournalProceedings of the National Academy of Sciences of the United States of America
Issue number7
StatePublished - Feb 15 2011
Externally publishedYes


  • Ciliary length
  • Eye development
  • Intraflagellar transport
  • Microtubules

ASJC Scopus subject areas

  • General


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