ZFIN ID: ZDB-PUB-140402-20
C2orf62 and TTC17 Are Involved in Actin Organization and Ciliogenesis in Zebrafish and Human
Bontems, F., Fish, R.J., Borlat, I., Lembo, F., Chocu, S., Chalmel, F., Borg, J.P., Pineau, C., Neerman-Arbez, M., Bairoch, A., and Lane, L.
Date: 2014
Source: PLoS One   9(1): e86476 (Journal)
Registered Authors: Bontems, Franck, Fish, Richard, Lane, Lydie, Neerman-Arbez, Marguerite
Keywords: none
MeSH Terms:
  • Actins/physiology*
  • Animals
  • Base Sequence
  • Carrier Proteins/genetics
  • Carrier Proteins/metabolism*
  • Cell Line
  • Cilia/genetics
  • Cilia/physiology*
  • Computational Biology
  • DNA Primers/genetics
  • Fluorescence Resonance Energy Transfer
  • Gene Expression Profiling
  • Gene Knockdown Techniques
  • Green Fluorescent Proteins
  • Humans
  • Immunohistochemistry
  • In Situ Hybridization
  • Luminescent Proteins
  • Real-Time Polymerase Chain Reaction
  • Reverse Transcriptase Polymerase Chain Reaction
  • Sequence Alignment
  • Two-Hybrid System Techniques
  • Zebrafish/genetics*
PubMed: 24475127 Full text @ PLoS One

Vertebrate genomes contain around 20,000 protein-encoding genes, of which a large fraction is still not associated with specific functions. A major task in future genomics will thus be to assign physiological roles to all open reading frames revealed by genome sequencing. Here we show that C2orf62, a highly conserved protein with little homology to characterized proteins, is strongly expressed in testis in zebrafish and mammals, and in various types of ciliated cells during zebrafish development. By yeast two hybrid and GST pull-down, C2orf62 was shown to interact with TTC17, another uncharacterized protein. Depletion of either C2orf62 or TTC17 in human ciliated cells interferes with actin polymerization and reduces the number of primary cilia without changing their length. Zebrafish embryos injected with morpholinos against C2orf62 or TTC17, or with mRNA coding for the C2orf62 C-terminal part containing a RII dimerization/docking (R2D2) – like domain show morphological defects consistent with imperfect ciliogenesis. We provide here the first evidence for a C2orf62-TTC17 axis that would regulate actin polymerization and ciliogenesis.