LAUSR

present in metabolism, differentiation, and development. Derangement of ciliary structures and activities underlies the pathogenesis of severe genetic disorders,alsoknownasciliopathies.Theprimaryciliumisformeddur-ing G1 phase of the and disassembled at the G2/M phase transition. Dynamics of the primary cilium is finely regulated at mul-tiple levels by the ubiquitin-proteasome system (UPS). Components of the UPS, including E2 ubiquitin-conjugating enzymes, E3 ubiquitin ligases and deubiquitylating enzymes (DUBs) identified as resident of—or closely associated to—the ciliary compartment. Here, the ubiquitin-directed proteolysis of key substrates is functionally relevant for cilium dynamics . The by Habeck and Schweiggert describes the current of the role of the in primary ciliogenesis, with a special focus on the timing of ubiquitylation events underlying cilium formation and disassembly. Evidence indicate that the can either support or inhibit the formation of the cilium, mostly depending on and when the ubiquitin