The pathophysiology of dystonia is incompletely understood. Unlike many other focal dystonias, cervical dystonia, a frequent dystonia leading to twisting of the head, does not appear to be related to overuse or acquisition of a demanding motor skill. Here, we report development of task-specific dystonia of the neck muscles in a 67-year-old patient following bilateral traumatic arm amputation at the age of 15. To compensate for the amputation, the patient learned to write with a pen held in his mouth. After several years of practicing this unusual and demanding skill, symptoms of task-specific cervical dystonia (CD) developed. This dystonia later became permanent, and independent of the motor activity that initially triggered the dystonic muscular contractions. This singular case raises the possibility that the pathophysiology of CD may share common elements with that of focal dystonias in different body regions.