Of note, DRD2 and RGS9‐2 protein expression levels were also reduced in Tor1aΔgag/+ striatum (Fig 1B; DRD2: Tor1a+/+ 1.000 ± 0.123 N = 7, Tor1aΔgag/+ 0.648 ± 0.067 N = 9, t‐test P = 0.0183; RGS9‐2: Tor1a+/+ 1.000 ± 0.123 N = 10, Tor1aΔgag/+ 0.556 ± 0.119 N = 9, t‐test P = 0.0192), similarly to what we observed in Tor1a+/− mice, further strengthening the relevance of dopaminergic dysfunction in DYT1 dystonia. Here, DRD2 is linked to early-onset generalized limb-onset dystonia.