RNA editing restricts hyperactive ciliary kinases

Abstract

RNA editing restricts ciliary kinases Ciliary kinases are essential for cilia formation and function, but it remains unknown how their activities are regulated in vivo. Li et al . created roundworm animal models carrying hyperactive ciliary kinases that disrupt cilia. Their genetic suppressor screens revealed that loss of an RNA adenosine deaminase, which catalyzes adenosine-to-inosine (A-to-I) RNA editing, rescued ciliary abnormalities. They found that kinase hyperactivation caused this RNA adenosine deaminase to edit kinase RNA and impair kinase RNA splicing and translation, thereby downregulating ciliary kinases from nuclei. These results suggest that ciliopathies may be treated by targeting the pathways outside of cilia. —DJ