Background Buschke–Löwenstein tumor is a giant condyloma acuminata infection that is characterized by degeneration, invasion, and recurrence. It is associated with human papilloma virus infection. It develops around the genital and perineal area, sometimes causing a large budding ulcerated lesion. Although human immunodeficiency virus infection is frequent in Africa, there are few descriptions of Buschke–Löwenstein tumor diagnosis and its management. Screening for other sexually transmitted infections must be systematic among these patients. Case presentation We report herein the case of a 21-year-old African origin male patient who developed a perineal swelling. Physical examination showed evidence of a huge exophytic tumor made up of budding pinkish vegetations, with serrated crests, a ‘’butterfly wing’’ structure, and a cauliflower-like appearance crowned with centrifugal circinate lesions. Multiple condylomatous lesions of the anal margin were also present. The patient tested positive for human immunodeficiency virus (cluster of differentiation 4 count of 119 cells/mm3) and hepatitis B infections. Real-time polymerase chain reaction revealed human papilloma virus-16 and other high-risk human papilloma virus deoxyribonucleic acid. The diagnosis of Buschke–Löwenstein tumor was made on mass biopsy, and the patient underwent multidisciplinary intervention (surgery, podophyllin application, and antiretroviral therapy). Medium-term evolution was, however, fatal due to opportunistic infection. Conclusion Buschke–Löwenstein tumor is a rare tumor associated with human immunodeficiency virus infection. It is more frequent in male human immunodeficiency virus-positive patients. There is a need to screen for other sexually transmitted infections. In most cases, the treatment is surgical, in association with local therapies. However, recurrences are common.