Severe dystonic tics involving the neck may lead to the development of serious spine disease. The pharmacological treatment of tics offers symptomatic relief, but clinically relevant improvement of severe tics is not frequently achieved and serious adverse events may result. Botulinum toxin (BnT) appears to be a safe and effective treatment for tics, but some concern exists about which group of patients may benefit from treatment. We report a patient affected by Tourette syndrome with tetraparesis and cervical myelopathy secondary to violent dystonic tics involving the neck, in which a more aggressive course of treatment with BnT in addition to neuroleptic medication resulted in complete resolution of cervical tics after 12 months of follow-up.