Bowen's disease, or squamous cell carcinoma in situ (SCCIS) of the skin, is a malignant neoplasm restricted to the epidermis, without evidence of dermal invasion. It usually develops in sun-exposed area of skin, but other sites can also be affected. Bowen's disease of the nipple is extremely rare and has thus far been reported only in women. We present the case of Bowen's disease of the nipple in an HIV-positive male patient who presented with a scaly lesion on nipple for one year. He also had past genital infection with human papillomavirus, but he was found to be negative for high-risk subtypes. Biopsy of the lesion revealed SCCIS of the nipple areola complex, with extension into the underlying lactiferous ducts of the breast. There was no evidence of invasive carcinoma. The patient was treated with a simple mastectomy with sentinel lymph node biopsy. With the advent of highly active antiretroviral treatment, chronic non-HIV related conditions have become more important, male breast cancer being one of them. To the best of our knowledge, this is the first reported case in the worldwide literature of Bowen's disease of the nipple in a young immunocompromised male patient. More aggressive therapy in HIV-positive male patients presenting with precancerous and cancerous breast lesions is recommended.