Manganese plays an important role as a cofactor in many enzymatic reactions in humans but in excess amounts can cause irreversible nervous system damage. Although manganism is a rare condition, it can be the cause of complex nervous system symptoms, especially in the setting of environmental exposure. Specifically, manganese is a well-known cause of dystonic parkinsonism. This article highlights several historical descriptions of the clinical manifestations, pathological changes, and attempted therapeutic intervention in manganese intoxication.