Functional analysis of dopaminergic systems in a DYT1 knock-in mouse model of dystonia

► Subtle abnormalities in motor skills were found in a DYT1 knock-in model of dystonia with multiple motor tasks. ► Striatal microdialysis showed reduced baseline and amphetamine-stimulated dopamine release. ► Anatomical studies revealed a small reduction in TH-immunopositive nigrostriatal neurons, with a small increase in soma size.