The role of pathogenic mutations in ADCY5 was first recognized in 2012, when a segregating missense change in the gene was discovered in a large dominant kindred with multiple affected members presenting with an early-onset hyperkinetic movement disorder named Familial Dyskinesia with Facial Myokymia (FDFM; OMIM 600293) [1], [2]. Here, ADCY5 is linked to Familial dyskinesia and facial myokymia.