Our work is focused on malformations of the face and cranium, particularly the premature fusion of the joints (“sutures”) between the bones of the skull vault (this is called craniosynostosis). A web of developmental mechanisms is involved in organising this complex system of bones, and a variety of genetic mutations can affect these processes to cause serious skull malformations. The talk will move from the identification of new genetic causes of craniosynostosis, through modelling in mice and induced pluripotent stem cell (iPSC) lines to establish causality and investigate pathogenic mechanisms, to a discussion of what the human and mouse genetics tell us about normal cranial suture and skull development.