Boceprevir and telaprevir are 2 specific inhibitors of the hepatitis C (HCV) serine protease 3. Cutaneous side effects have been reported with high frequency, essentially rash, and dry skin. We report a case of drug rash with eosinophilia and systemic symptoms (DRESS) due to boceprevir. A 56-year-old African woman with chronic hepatitis C complicated with cirrhosis and cryoglobulinemia received pegylated interferon alfa-2a (PegIFN) and ribavirin (RBV) for 4 weeks and then boceprevir was added. She was also co-infected with HIV state A2. Eight weeks after adding boceprevir she developed a generalized maculopapular exanthema with fever, facial oedema, apparition of lymph node and alteration of the general state. She presented an eosinophilia (up to 3.0 × 10(9)cells/L), no biological inflammatory syndrome. The computed tomography revealed several lymph nodes located in the abdominal and inguinal areas. The cutaneous biopsy was consistent with a drug rash reaction. The HCV treatment was stopped and the patient was treated with topical steroids. Cutaneous and systemic symptoms disappeared in few weeks. Boceprevir was considered the culprit drug. We report to our knowledge the first case of DRESS due to boceprevir.