Structural lesions of CNS, reported to be associated with torticollis, are mostly restricted to cerebellum, brain stem and basal ganglia. In fact, we know only about two documented frontal lobe mass lesions—meningiomas, associated with torticollis. Our observation of frontal lobe cavernous angioma associated with clinical picture of torticollis confirms the role this area could play in the pathophysiology of involuntary movements. We report a case of patient with torticollis associated with cavernous angioma of the right frontal lobe and presuppose causative role of angioma in the development of our patient’s torticollis.