Domperidone is a dopamine receptor antagonist with gastrokinetic and anti-emetic effects. Domperidone is widely used worldwide as an anti emetic and in the treatment of gastroparesis and gastroesophageal reflux. Because domperidone does not readily cross the blood-brain barrier, central nervous system side effects are not expected. Herein, we reported a 12-year-old girl who presented with dystonia after domperidone usage. We present a 12-year-old girl with a 40 kg body weight who was admitted to our pediatric emergency department with numbness, stiffness, and twisting in the neck, hands, arms, and tingling in the legs and back. We administered 3 mg biperiden, an anticholinergic, through slow intravenous infusion. The patients dystonic reaction recovered 10 min after biperiden infusion. Based on our literature search, we presented one of the few acute dystonia cases after domperidone use in children. To the best of our knowledge, our case is the first case report in children who were successfully and rapidly treated with anticholinergic, biperiden.Keywords: Biperiden, cholinergic antagonists, domperidone, dopamine antagonist, dystonia.