Drug-induced hypersensitivity syndrome (DHS), also named drug rash with Eosinophilia and systemic symptoms (DRESS) is a potentially dangerous side effect of some drugs, especially antiepileptic drugs (AEDs) such as phenobarbital, phenytoin, carbamazepine, lamotrigine, primidone, etc. It can also be caused by other drugs, such as sulfonamides and captopril. Diagnosis of DHS may be difficult because of the variety of clinical and laboratory abnormalities and manifestations and because the syndrome may mimic infectious or collagen vascular disorders. Management includes immediate withdrawal of the culprit drug, supportive care and systemic steroids or Immunoglobulins (IVIG). Here, we briefly reviewed the literature, followed by a case report that had all of the criteria of DRESS without eosinophilia.