Phosphoinositides are phospholipids that regulate signal transduction,
endocytosis, and protein trafficking. Each phosphoinositide has a unique pattern of
distribution within cellular compartments and is tightly regulated by inositol kinases
and phosphatases localized within a specific membrane compartment. Inositol
polyphosphate 5-phosphatases regulate phosphoinositide levels and are implicated in
human diseases, such as Lowe syndrome and Joubert syndrome. Here we review the
pertinent findings of the roles of 5-phosphatases in cilia function and signaling.
Keywords: Cilia, INPP5E, Joubert syndrome, Lowe syndrome, OCRL.
