Through its first two CAP‐Gly domains, CYLD interacts with microtubules and promotes the dynamics and stability of microtubules.[12, 15] Previously, we demonstrated that CYLD participates in RhoA‐mediated cytoskeletal rearrangement by deubiquitinating leukemia‐associated RhoGEF.[22] In this study, we found that the deubiquitinase activity of CYLD contributes to RPE microvillar assembly and POS phagocytosis. This evidence concerns the gene RHOA and leukemia.