Motor tics are brief, repetitive, involuntary movements that interfere with behavior and appear in multiple neural disorders, most notably, Tourette syndrome. Converging evidence from different lines of research point to the involvement of the corticobasal ganglia system in tics, but the neural mechanism underlying motor tics is largely unknown. An animal model directly linking basal ganglia dysfunction and motor tics indicated that local disinhibition within the basal ganglia input structure, the striatum, induces the appearance of motor tics in both rats and monkeys. Recordings of neuronal activity from multiple brain regions performed in this model during the expression of motor tics showed that tics are associated with phasic changes of neuronal activity throughout the corticobasal ganglia pathway, culminating in the disinhibition of the cortex and the release of a tic. This line of research provides a mechanistic description of the underlying neurophysiology of motor tics and may supply the much needed infrastructure for methodical hypothesis-driven studies of novel clinical treatments.