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Infantile-Onset Paroxysmal Dystonia: A Diagnostic Dilemma

Department of Neurology, The New Children's Hospital Sydney, Australia

Robert Howman-Giles, MD, FRACP

Department of Nuclear Medicine, The New Children's Hospital Sydney, Australia

Jayne Antony, MD, FRACP

Department of Neurology The New Children's Hospital Sydney, Australia

A 4-year-old boy presented with a history of paroxysmal dystonic posturing since birth. Episodes were triggered by stress, fatigue, and cold. Sleep, for as short as 1 minute, resulted in complete resolution of dystonia. He was developmentally normal, with no focal neurologic deficits. Cerebrospinal fluid, homovanillic acid (HVA), and 5-hydroxyindoleacetic acid (5-HIAA) were borderline low. On ictal spectroscopy, there was reduced blood flow to the right temporal region, caudate nuclei, and thalami. The typical infantile form of dystonia is benign, resolving by 2 years of age in an otherwise normal child. Our patient remains symptomatic at 4 years of age. (J Child Neurol 2001;16:222-225).

Journal of Child Neurology, Vol. 16, No. 3, 222-225 (2001)
DOI: 10.1177/088307380101600312


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