Infantile-onset paroxysmal dystonia: A diagnostic dilemma

A 4-year-old boy presented with a history of paroxysmal dystonic posturing since birth. Episodes were triggered by stress, fatigue, and cold. Sleep, f or as short as 1 minute, resulted in complete resolution of dystonia. He wa s developmentally normal, with no focal neurologic deficits. Cerebrospinal fluid, homovanillic acid (HVA), and 5-hydroxyindoleacetic acid (5-HIAA) wer e borderline low. On ictal spectroscopy, there was reduced blood flow to th e right temporal region, caudate nuclei, and thalami. The typical infantile form of dystonia is benign, resolving by 2 years of age in an otherwise no rmal child. Our patient remains symptomatic at 4 years of age.