FIGURE 6:

Schematic depicting ccp5 knockdown–induced changes to tubulin glutamylation in normal and flee-deficient cilia. In normal cilia of wild-type single ciliated cells (SCCs) and MCCs, glutamylated tubulin is enriched maximally at the base and gradually decreases toward the tip. Loss of ccp5 in otherwise normal cilia significantly increases the overall amount and extent of glutamylated tubulin without increasing cilia length. Hyperglutamylation inhibits efficient cilia motility. Glutamylated tubulin is significantly reduced and restricted to the base of short, single cilia that persist in the fleer mutants. Fleer-deficient cilia remain competent for tubulin glutamylation and show greater-than-normal increase in overall intensity and extent when Ccp5 deglutamylase is knocked down in the fleer mutants. Multicilia in fleer mutants are more sensitive to the loss of glutamylation; however, multiciliated cells in compound fleer/ccp5-deficient zebrafish show improved assembly of cilia upon increase in tubulin glutamylation.