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<p><strong>Background:</strong> A number of neurological conditions have been reported to be associated with gluten sensitivity, including ataxia, peripheral neuropathy, epilepsy, and occasionally, chorea. The pathogenic role of anti-gliadin antibodies has been questioned, and pathophysiology remains controversial.<br /><br /><strong>Case Report:</strong> I report chorea in a patient with celiac disease, which responded to a gluten-restricted diet. The response of the movement disorder to change in diet strongly suggests a functional role for anti-gliadin antibodies in the generation of chorea, probably involving basal ganglia targets.<br /><br /><strong>Discussion:</strong> Gluten sensitivity may be a treatable cause of chorea.</p>