Tardive dyskinesia (TD) is a dreaded side effect of antipsychotic medication. Recommended treatments for TD may provide reliable improvement but can be, in turn, associated with additional adverse reactions. Recently, several reports have suggested that botulinum toxin A (BTX-A) injection in affected muscles may significantly improve TD. Here, we report a case of severe tongue protrusion dystonia secondary to an antipsychotic medication in a young man. Several approaches including clozapine, amisulpride, aripiprazole, ziprasidone, tiapride and clonazepam failed to improve the symptoms. Injection of 50 U of BTX-A (Dysport, Ipsen, Ettlingen, Germany) into each genioglossal muscle dramatically improved tongue protrusion within few days with a sustained effect. If reasonable precautions are taken, the application seems to be well tolerated with only minor side effects. A review of the literature that is part of this article adverts BTX-A injection as a potential beneficial approach of various kinds of TD.