Extrapyramidal disorders are a rare presentation of multiple sclerosis. One such presentation is paroxysmal hemidystonia. Dystonia is, in the strict sense, the result of damage to various sites in the central nervous system. In one published cases series, lesions causing dystonia occurred in heterogeneous locations, most commonly in the basal ganglia, followed by the thalamus, brainstem, and white matter. In a series of 62 patients with thalamic or peritalamic lesions, only about 5% patients had paroxysmal dystonia. So far, there have been few reported cases of patients with paroxysmal hemidystonic symptoms associated with a specific damage to the central nervous system in the course of multiple sclerosis, especially as a relapse. For these reasons, we present a case of a 57-year-old female patient with a medical history of about 30 years of multiple sclerosis, who developed symptoms of paroxysmal left-sided hemidystonia as a relapse of multiple sclerosis.

movements disorders, paroxysmal hemidystonia, thalamic and subthalamic lesions, multiple sclerosis, relapse