The treatment of malocclusions can involve orthognathic surgery. Although orthognathic surgery is a safe surgical procedure, low incidences of many complications have been reported such as infections, hemorrhage, nerve injuries, temporomandibular disorders (TMDs), and psychological problems. There are no reports in the literature of orthognathic surgery being associated with postural disorders although the link between dental occlusion and postural disorders is highlighted in numerous recent publications. This report describes the case of a young, healthy patient who presented with sustained spasmodic torticollis following orthognathic surgery. In addition, the physiopathological aspects of this atypical condition are discussed.