Drug rash with eosinophilia and systemic symptoms (DRESS) syndrome, is an uncommon, acute and severe life-threatening systemic disease. DRESS syndrome is characterised by fever, skin rash, lymph node enlargement, and internal organ involvement. The most common culprit drugs are anticonvulsants and allopurinol. We first reported a case of DRESS syndrome induced by cefazolin and gentamicin in a child. We aimed to emphasize the severity of renal damage and the dramatic response to short term intravenous and oral steroid therapy.