Ciliogenesis is the culmination of a succession of spatially and temporally coordinated steps that may differ from one organism to another. However, it includes several key steps, common to all cellular models and conserved throughout evolution: duplication of the centriole/basal body, maturation of the basal body, migration and anchoring to the membrane leading to the formation of the transition zone and then growth of the cilium. In addition to a correct synthesis of the proteins involved, the assembly of this complex organelle depends on the correct protein-protein interactions in a 4-dimensional space-time and on their regulations by post-translational modifications such as ubiquitination/deubiquitination. Cilia are involved in many developmental and physiological processes and their alteration, in mammals, leads to physio-pathological disorders called “ciliopathies“. Otherwise, the loss of cilia observed in various tumors, reveals that ciliogenesis defects could be involved in some cancers.