Of note, the critical role of PDE10A in the control of striatal-dependent movement is demonstrated by mutations in the coding sequence of the PDE10A gene that cause infantile-onset limb and orofacial dyskinesia (IOLOD, OMIM #616921), in which there is a reduction of the protein in the striatum [74], and autosomal dominant striatal degeneration-2 (ADSD2, OMIM # 616922), in which the activity of PDE10A is affected, more precisely the binding to cAMP to stimulate the PDE10A catalytic domain [75]. This evidence concerns the gene PDE10A and infantile-onset generalized dyskinesia with orofacial involvement.