DRESS (Drug reaction with eosinophilia and systemic symptoms) syndrome is a rare, potentially life-threatening, drug induced hypersensitivity reaction manifested by fever, rash, eosinophilia, lymphadenopathy, and organ involvement especially liver and kidney. The disease is characterized by a long latency period (at least two weeks) between the drug exposure and disease onset. The most commonly reported drugs associated with DRESS syndrome in the literature are allopurinol, and anticonvulsants. We describe a patient presented with eosinophilia, fever, diffuse maculopapular rash, hepatomegaly, and multiple intra-abdominal lymphadenopathies just ten days after initiation of cefuroxime axetil. In our case, we aim to announce the first case report of cefuroxime axetil related DRESS syndrome, and also speculate on the possible association between cephalosporin and DRESS syndrome.