PUBLICATION

C2orf62 and TTC17 Are Involved in Actin Organization and Ciliogenesis in Zebrafish and Human

Authors
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.
ID
ZDB-PUB-140402-20
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
Abstract

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.

Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping