The aim of this study was to verify whether botulinum toxin (BTX)-induced clinical improvement of cranial dystonia is associated with changes in the cortical silent period (SP), a measure of cortical excitability. By transcranial magnetic stimulation (TMS), high-intensity stimuli were delivered with a round coil centered at the vertex during a maximal muscle contraction of the orbicularis oculi. Motor evoked potentials (MEPs) and SPs were obtained from surface electrodes placed over the orbicularis oculi muscle before and 2 to 3 weeks after BTX-A injection into the affected muscles in 10 patients with cranial dystonia and 8 age-matched control subjects. BTX injection improved blepharospasm in all patients. Facial muscle SPs were significantly shorter in patients than in control subjects and did not significantly change after treatment, at the time of maximal clinical improvement. We conclude that the clinical improvement induced by BTX in patients with cranial dystonia is largely symptomatic. It does not appear to result from modulation of abnormal aspects of intracortical excitability, although these may play a role in craniofacial dystonia.