Congenital muscular torticollis results from shortening or excessive contraction of the sternocleidomastoid (SCM) muscle. The reported incidence varies between 0.4 and 1.9%. Various theories have been proposed, but its true aetiology remains obscure. The deformity is characterized by a practically painless, contracted cordlike SCM muscle, which pulls the head toward the side affected, narrows and draws the shoulder upward, forcing the chin in the opposite direction. Torticollis of congenital origin is a deformity not commonly met with in the practice of maxillofacial surgery. The rarity, lack of, or inadequacy of the previous treatment, together with the advanced age and marked deformity appear to warrant an investigation and report of the outcome. A case of congenital muscular torticollis is presented who reported at the age of 18 years and has been successfully treated by unipolar SCM release.