Pisa syndrome (PS) is characterized by an abnormally sustained posture with flexion of the body and head to one side and slight axial rotation of the trunk. PS has been related to the use of neuroleptics and is clinically classified as either an acute or a tardive dystonia. This is the first report describing a case of late-onset PS, occurring in a young patient treated with aripiprazole for 2 years. To establish optimal treatment management, we reviewed the literature on aripiprazole-induced PS and tardive dystonia. In light of current knowledge, we proposed a multistep algorithm to aid in the clinical management of this condition.