A novel antiepileptic drug, levetiracetam, has been reported to cause several psychiatric adverse effects in spite of its effectiveness on epilepsy. However, a possible relationship between levetiracetam and obsessive-compulsive behavior has only been reported in a few studies with adult epilepsy patients. We treated a pediatric patient with epilepsy without past or family history of psychiatric disorder. Levetiracetam was started to control generalized tonic-clonic seizure. Two months after initiation of levetiracetam with favorable seizure control, she started to show an obsessive-compulsive behavior such as repetitive checking of her back, pants, and chair. Based on the course of its appearance, levetiracetam administration was identified as a possible cause. After termination of levetiracetam, her obsessive-compulsive behavior completely disappeared with reappearance of seizures. This case provides clear evidence that levetiracetam may cause obsessive-compulsive behavior even in a pediatric epilepsy patient without psychiatric background, possibly mediated by modulation of the glutamate system by levetiracetam.
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