In the present work, we investigated the molecular mechanisms underlying DRD2 reduced levels and altered signaling in the striatum of DYT1 dystonia models, Tor1a+/−‐knock‐out and Tor1a∆gag/+‐knock‐in mice (Goodchild et al, 2005). Here, DRD2 is linked to early-onset generalized limb-onset dystonia.