BackgroundBuschke–Lowenstein (B–L) tumors are rare, aggressive warty lesions that can grow to enormous sizes and have a risk of malignant transformation despite their benign histology. These tumors are primarily caused by HPV types 6 and 11, with additional risk factors, including immunodeficiency, multiple sexual partners, poor hygiene, and a history of other genital infections. The typical treatments include topical medications, cryotherapy, chemotherapy, radiotherapy, and surgery. Surgery is considered the gold standard, particularly for extensive and infiltrative lesions, as demonstrated in our case.Case presentationWe report a case of a male patient with perianal and penopubic B–L tumors, who had multiple sexual partners and no history of homosexual relationships, presenting with refractory classic condyloma. The perianal tumor exhibited malignant transformation, while the penopubic tumor remained histopathologically benign. Upon admission, the patient showed unusual biochemical blood results, including hypercalcemia and anemia of chronic illness. The operation and postoperative period were managed by a multidisciplinary team. The patient was satisfied with both the cosmetic and functional outcomes. Histopathological examination confirmed the suprapubic lesion as a B–L tumor, whereas the perianal specimen revealed a small focus of squamous cell carcinoma arising from a B–L tumor.ConclusionsWith an experienced multidisciplinary team, excision of the tumor is still the standard treatment for BL tumor. Achieving a negative surgical margin for any SCC foci is of utmost importance to prevent recurrence. Close and careful follow-up is essential due to the high recurrence rate associated with these tumors. Further research is needed to clarify existing controversies and to enrich the literature on this rare phenomenon.