An anticholinergic reverses motor control and corticostriatal LTD deficits in Dyt1 ΔGAG knock-in mice

► Anticholinergic trihexyphenidyl restored long-term depression in Dyt1 ΔGAG mice. ► Trihexylphenidyl also reversed motor control deficit in dystonia mouse model. ► Striatal D2 receptors were expressed at lower quantities in Dyt1 ΔGAG mice. ► Ca2+ channel agonist FPL64176 only partially induced dystonic signs in mutant mice. ► Cholinergic tone is important in synaptic plasticity and motor control in dystonia.