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A 54-year-old woman developed tardive tongue dyskinesia and burning mouth syndrome approximately 5 years after starting antipsychotic [neuroleptics] therapy [drug details and therapeutic indication not stated]. She had involuntary, disabling, choreic tongue movements that were described as continuous, irregular and writhing during mouth closing, plus severe mouth burning symptoms. She was treated with 50 units of botulinum toxin A injected into the tongue muscles. Around 10 days after the injection, the tongue dyskinesia and burning symptoms had improved. She received botulinum toxin A every month for 2 years after the initial injection.