Abstract
Nocturnal Paroxysmal Dystonia (NPD) is characterized by abnormal dyskinetic movements occur mostly during non-REM sleep. From introducing NPD in early 1980s, most authors have been in favour of an epileptic origin for these sleep-related episodes. Clinical characteristics of NPD including abrupt hypermotor behaviours, automatisms and vocalizations, abnormal EEG findings in some patients and therapeutic response to anti-epileptic drugs persuade clinicians to accept NPD as sleep-related epilepsy with frontal lobe origin but it seems this conclusion may not be true in all cases. We present a young adult patient with refractory NPD who responded to antihistamine cyproheptadine and propose an alternative theory to describe NPD according to the basal ganglia dysregulation. So, in such patients, other therapeutic approach should be reasonably sought.
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