Congenital muscular torticollis (CMT) is the most common cause of torticollis in childhood. This condition is usually recognized and successfully treated in infancy, but may persist in adulthood, particularly if not treated. In adult patients, CMT can be differentiated from idiopathic cervical dystonia by the frequent association with facial asymmetry, presence of a cord-like sternocleiodmastoid muscle (SCM), absence of head tremor, lack of sensory trick, and head tilt since infancy. We describe 3 patients with persistent CMT, who were successfully treated with botulinum toxin injections with long lasting benefit. © 2010 Movement Disorder Society.