SummaryWe report a case of cranial dystonia related to the administration of ranitidine. The symptoms started shortly after institution of treatment, resolved after discontinuation, and recurred upon repeat administration of the drug. Although there have been a few reports of abnormal involuntary movements related to other histamine2 antagonists, this is only the second reported instance due to ranitidine and the first reported instance of dystonia related to the drug. The pathophysiology of this effect is unclear, but a central cholinergic effect of this agent may be a contributing factor.