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Since all neuroleptics (antipsychotics) share the ability to block the dopamine D<jats:sub>2<\/jats:sub>-type receptors (D<jats:sub>2<\/jats:sub>Rs) that are highly enriched in the striatum, this drug-induced dystonia is thought to be caused by decreased striatal D<jats:sub>2<\/jats:sub>R activity. However, how associations of striatal D<jats:sub>2<\/jats:sub>R inactivation with dystonia are formed remains elusive.<\/jats:p><jats:p>A growing body of evidence suggests that imbalanced activities between D<jats:sub>1<\/jats:sub>R-expressing medium spiny neurons and D<jats:sub>2<\/jats:sub>R-expressing medium spiny neurons (D<jats:sub>1<\/jats:sub>-MSNs and D<jats:sub>2<\/jats:sub>-MSNs) in the striatal striosome-matrix system underlie the pathophysiology of various basal ganglia disorders including dystonia. Given the specificity of the striatal dopamine D<jats:sub>1<\/jats:sub>system in \u2018humans\u2019, this article highlights the striatal striosome hypothesis in causing \u2018repetitive\u2019 and \u2018stereotyped\u2019 motor symptoms which are key clinical features of dystonia. It is suggested that exposure to neuroleptics may reduce striosomal D<jats:sub>1<\/jats:sub>-MSN activity and thereby cause dystonia symptoms. This may occur through an increase in the striatal cholinergic activity and the collateral inhibitory action of D<jats:sub>2<\/jats:sub>-MSNs onto neighbouring D<jats:sub>1<\/jats:sub>-MSNs within the striosome subfields. The article proposes a functional pathology of the striosome-matrix dopamine system for neuroleptic-induced acute dystonia or neuroleptic-withdrawal dystonia. A rationale for the effectiveness of dopaminergic or cholinergic pharmacotherapy is also provided for treating dystonias. 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