The motile cilium in development and disease: emerging new insights.

In this paper, I review a collection of recently published papers that have provided significant new information about the biogenesis and functions of motile cilia. In vertebrates, the activity of motile cilia has been associated with a fascinating diversity of developmental and physiological processes. Despite the importance, much remains to be learned about the genetic control and cellular events that are involved in the differentiation of motile cilia. We also need to better understand the mechanisms by which cilia-driven fluid flow is able to influence such a variety of developmental and physiological processes. The Foxj1 family of proteins has now been definitively established as master regulators of motile ciliogenesis.1,2 Identification of the Kintoun/PF13 protein has shed light on the assembly of dynein arms,3 whereas live imaging of ciliary motility has led to the discovery of an intriguing new role for motile cilia in otolith formation in the ear.4.