AbstractIntroduction: DRESS (drug reaction with eosinophilia and systemic symptoms) syndrome is a potentially life-threatening illness with cutaneous and systemic manifestations, following exposure to an offending drug (like anti-convulsants, antimicrobials or anti-inflammatory drugs). It presents with fever, rash, hematological abnormalities & internal organ involvement. Removal of the offending agent & use of systemic glucocorticoids are the .cornerstones of treatment Case Report: An eight-years-old boy, on treatment with phenytoin (for generalized tonic-clonic seizure which had occurred 8 weeks ago), presented with- fever, rash, periorbital swelling & pedal edema. Examination revealed bilateral cervical lymphadenopathy, pedal edema & erythematous maculopapular rash (on abdomen, back, upper & lower limbs) with peeling & erosions over the lips & angle of mouth. Blood investigations showed leukocyte count of 13,600 cells/cumm with eosinophilia (absolute count- 1,632/cumm), elevated liver transaminases (SGOT- 146 IU/L; SGPT- 78 IU/L) & renal failure (elevated BUN- 18 mg/dl & creatinine- 1.1 mg/dl). Renal biopsy suggested interstitial nephritis. Phenytoin was discontinued on suspicion of DRESS syndrome. Patient was treated with oral steroids & hemo-dialysis (for the rising creatinine). Anti-epileptic drug was changed to levetiracetam. The patient recovered in 4 weeks. Parents were counselled about risk of reaction to similar drugs & advised regular follow-up to watch .for relapse Conclusion: DRESS syndrome (although rare in children) should be considered, especially if history of recent addition of potentially incriminated drugs exists. Most patients with DRESS recover completely .in few weeks to months after discontinuing the offending drug