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Drug-induced Pisa syndrome (DIPS) or pleurothotonus, characterized by tonic şexion of the trunk and head to one side and slight rotation of the body. Pisa syndrome occurs mainly during medication with typical, but sometimes also with atypical antipsychotics. The pathophysiology of Pisa syndrome is complex; a dopaminergic-cholinergic imbalance or serotonergic or noradrenergic dysfunction might be a possible explanation. Some cases of induction of the condition by neuroleptics and improvement with anticholinergic agents may suggest that a dopaminergic-cholinergic imbalance might be the main factor. Aripiprazole is an atypical antipsychotic, acting as partial agonist at dopamine D2 and serotonin 5-HT1A receptors and as an antagonist at 5-HT2A. Aripiprazole appears to be associated with low extrapyramidal side effect (EPS) rates in pediatric patients. Aripiprazole may alter the dopaminergic–cholinergic balance, which is supposed to be the main underlying pathogenetic factor in Pisa syndrome. But the underlying pathophysiological mechanisms of DIPS are still unclear. In the literature, there was no DIPS condition in pediatric population following the psychiatry treatment. We report a case of Pisa Syndrome caused by short term use of aripiprazole in an autistic child. A 12-years-old male child with autism had been treated with aripiprazole 5 mg/day for aggression during the last 2 months. When the dose of aripiprazole was increased from 5 mg to 7.5 mg the patient demonstrated a tilting of his trunk toward right side. Within a couple of days of being taken off the treatment, the patient no longer exhibited the symptoms of Pisa syndrome. As indicated by previous reports, reduction in dose or discontinuation of the antipsychotic drug remains to be the first-line treatment for Pisa syndrome. In some researches, it is suggested that aripiprazole could improve DIPS because of its partial dopamine agonist effects without complete blocking of dopaminergic activity in the nigrostriatal pathway. Physicians should be aware of Pisa syndrome in children and should alert patient of this possibility when starting and stepping up medications. The purpose of reporting this case is to create awareness among clinicians as it is a reversible condition, which responds to removal of the offending drug.