Levetiracetam (LEV) is a new antiepileptic drug (AED)that is effective in adults and children with partial-onset seizures or idiopathic or symptomatic generalized seizures. LEV does not bind to plasma proteins, and is eliminated by the kidneys. It has been argued that LEV can act on the N-type Ca 2? channel and can reverse the gammaaminobutyric acid (GABA) and glycine-gated currents [1]. Side effects include fatigue, somnolence, infection, headache, behavioral changes and skin rashes [2]. Herein, we present the first case of rhabdomyolysis in a child treated with LEV.A 13-year-old girl presented with a history of myalgia (mainly in her lower extremities) since last 2-3 days to our hospital. She was born of an uneventful full-term pregnancy. Developmental milestones were normal. On medical history, she had partial onset of secondarily generalized seizures during sleep since 2 months. The seizure activity consisted of twitching of the right face, tonic deviation of the mouth involving the lips, tongue, and pharyngeal and laryngeal muscles, resulting in speech arrest and drooling and progressing into bilateral tonic stiffening of the arms and legs. The EEG revealed left centrotemporal spikes and was subsequently diagnosed with benign epilepsy with centrotemporal spikes. The patient was administered LEV monotherapy. LEV had started 10 mg/kg/day, but her brief partial seizures continued. The dose was increased to 20 mg/kg/day (500 mg/day), and her seizures were ceased. One week after starting LEV therapy, she experienced mild myalgia. On her examination, the vital signs, including the blood pressure were normal. Her height and weight were in normal limit for her age. The physical and neurological examinations were normal. There was no evidence of trauma, exercise or infection.On laboratory examination, blood urea was 29 mg/dL (normal range 17-43), creatinine 1.0 mg/dL (normal range 0.6-1.2), myoglobin 78 ng/mL (normal range 14.3-65.8), and creatinine phospho kinase (CPK) was 986 U/L (normal range 27-168). Plasma carnitine (42 lmol/L; reference range 19-59 lmol/L) and free carnitine levels (35 lmol/L; reference range 12-46 lmol/L) were normal as acylcarnitine profile. Brain MRI showed no abnormal findings.Rhabdomyolysis was diagnosed with clinical and biochemical findings. We thought LEV may be the cause of her complaints and ceased LEV therapy. Hydration therapy were started to avoid potential complications. After stopping LEV, serum CPK and myoglobin levels gradually decreased and returned into normal values. Muscle biopsy was not performed.