Dystonia associated with antipsychotics is common and well known. The current case presents a 20-yearold male patient during the manic phase of bipolar disorder, developing piriformis syndrome (PS) under antipsychotic medication treatment. Initially, the anteflexion position of the patient was considered to be dystonia secondary to antipsychotic treatment and treated accordingly with biperiden, benzodiazepine, baclofen, tetrabenazine, electroconvulsive therapy (ECT), and botox; however, the patient did not respond to these treatments. Subsequently, after consulting the algology department, PS was diagnosed as a distinct entity. The patient recovered from the symptoms of PS upon the injection of bupivacaine and triamcinolone into his piriformis muscle. This case reminds us that the accuracy of the diagnosis in the treatment of possible drug-related side effects should be carefully reviewed, especially in resistant cases.