Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis are two rare but life-threatening diseases characterized by detachment of epidermis, bullous skin lesions and mucous membrane erosions. Anti-epileptic drugs are highly suspected to be the causative agents. Although carbamazepine (CBZ) is the most associated anti-epileptic drug, oxcarbazepine (OXC), which is a monohydrated derivative of CBZ, is proposed to be safer because of the different metabolism of the two drugs. Herein, we report a case of SJS induced by oxcarbazepine. A 6-year-old boy with benign rolandic epilepsy, admitted to our hospital with generalized maculopapular rash after starting oxcarbazepine. The diagnosis of SJS was made with cytotoxic skin lesions and mucous membrane involvement. After discontinuing of the drug and topical corticosteroid initiation, the lesions were improved. We report this case to attract attention to the serious side effect of this anti-epileptic drug.