Tic disorder, characterized by the presence of both motor and vocal tics is common in adolescents and adults. Antipsychotics including typical antipsychotics and atypical antipsychotics are generally recognized by experts as the most effective pharmacological treatment for tics. However, previous studies suggest that tic-like symptoms might manifest during treatment with atypical antipsychotics such as clozapine, quetiapine, but not aripiprazole. We present the first case, to our knowledge, of an adult schizophrenia patient who developed tics during treatment with aripiprazole.