Abstract: Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a severe drug-induced hypersensitivity reaction characterized by skin rash, fever, blood abnormalities, and multiple organ involvement. The diagnosis of DRESS syndrome is often delayed because of its variable presentation. DRESS syndrome induced by antituberculosis drugs has rarely been reported. A 45-year-old female patient taking anti-tubercular drugs since 2 weeks presented to our hospital with extensive skin lesion with mucosal involvement. The case responded well to treatment with systemic corticosteroids and withdrawal of isoniazid with no recurrence of lesions and further follow-up.