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Characterization of tetratricopeptide repeat-containing proteins critical for cilia formation and function

Yanan Xu1, Jingli Cao1, Shan Huang1

  • 1State Key Laboratory of Cell Biology, Institute of Biochemistry and Cell Biology, Shanghai Institutes for Biological Sciences, Chinese Academy of Sciences, 320 Yueyang Road, Shanghai, China.

Plos One
|April 11, 2015
PubMed

Insights

Tetratricopeptide repeat-containing (TTC) proteins are crucial for cilia function. This study identified four novel TTC genes essential for cilia formation and motility, revealing their roles in intraflagellar transport (IFT) and ciliopathies.

Area of Science:

  • Cell Biology
  • Genetics
  • Developmental Biology

Background:

  • Cilia are vital cellular structures requiring intraflagellar transport (IFT) machinery for assembly and function.
  • Tetratricopeptide repeat-containing (TTC) proteins are involved in protein complex formation, with nine previously linked to IFT or BBSome complexes.
  • The full extent of TTC protein involvement in cilia and their specific functions remain largely uncharacterized.

Purpose of the Study:

  • To investigate the role of Tetratricopeptide repeat-containing (TTC) proteins in cilia formation and function.
  • To identify novel TTC genes critical for cilia development and motility.
  • To elucidate the molecular mechanisms by which TTC proteins interact with the intraflagellar transport (IFT) machinery.

Main Methods:

  • Differential gene expression analysis of TTC genes during mouse tracheal epithelial cell (MTEC) differentiation.
  • Systematic genetic screening in zebrafish to identify TTC genes essential for cilia.
  • Phenotypic analysis of zebrafish morphants, including assessment of ciliopathy-related traits.
  • Co-immunoprecipitation assays to determine protein-protein interactions between TTC proteins and IFT/BBSome components.

Main Results:

  • Twenty TTC genes showed upregulation during MTEC differentiation into multiciliated cells.
  • Four novel TTC genes (ttc4, ttc-9c, ttc-36, ttc-39c) were identified as critical for zebrafish cilia formation and motility.
  • Zebrafish morphants exhibited characteristic ciliopathy phenotypes, such as hydrocephalus and abnormal left-right patterning.
  • TTC4 and TTC25 (a known cilia-related gene) morphants displayed pronephric cyst formation.
  • Immunoprecipitation confirmed interactions of TTC4, -9c, -25, -36, and -39c with IFT-A, IFT-B, or BBSome complexes.

Conclusions:

  • This study expands the known repertoire of cilia-related TTC proteins.
  • Identified TTC proteins are essential for cilia assembly, motility, and overall organismal development.
  • The findings highlight the significant role of TTC proteins in intraflagellar transport (IFT) pathways and associated ciliopathies.

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