Dystonias are characterized by involuntary muscle contractions, twisting movements, abnormal postures, and often tremor in various body regions. However, in the last decade several studies have demonstrated that dystonias are also characterized by sensory abnormalities. While botulinum toxin is the gold standard therapy for focal dystonia, exactly how it improves this disorder is not entirely understood. Neurophysiological studies in animals and humans have clearly demonstrated that botulinum toxin improves dystonic motor manifestations by inducing chemodenervation, therefore weakening the injected muscles. In addition, neurophysiological and neuroimaging evidence also suggests that botulinum toxin modulates the activity of various neural structures in the CNS distant from the injected site, particularly cortical motor and sensory areas. Concordantly, recent studies have shown that in patients with focal dystonias botulinum toxin ameliorates sensory disturbances, including reduced spatial discrimination acuity and pain. Overall, these observations suggest that in these patients botulinum toxin-induced effects encompass complex mechanisms beyond chemodenervation of the injected muscles.