Introduction:Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a severe, potentially life-threatening condition precipitated by reaction of therapeutic drugs. The prevalence of potential antitubercular therapy (ATT)-induced DRESS is 1.2%.Case presentation:A 71-year-old female patient after 5 weeks of starting ATT complaints of fever, vomiting, dizziness, and generalized itchy maculopapular rash over the body. It was associated with marked eosinophilia (absolute eosinophil count 3094 cell/mm3, 36% in peripheral blood smear).Discussion:Fever, rash, lymphadenopathy, and internal organ involvement with marked eosinophilia constitute the major clinical manifestations of DRESS. RegiSCAR scoring system is usually used to diagnose DRESS. Identification of the culprit drug is based on the temporal correlation of symptoms with drug exposure and rechallenge test, patch test and lymphocytic transformation tests may be valuable adjunctive tools. Treatment includes withdrawal of offending agent and use of topical or systemic corticosteroids, antihistamines, cyclosporin or JAK inhibitor with clinical judgement.Conclusion:Clinicians from the tuberculosis burden region must be aware of DRESS associated with ATT and they must counsel the patient properly before prescription and manage them without delay if DRESS ensues.