Trichoblastic carcinoma is a sporadic malignant tumor. It usually arises in the scalp, trunk, and extremities. However, its occurrence in the base of the penis is highly uncommon. To our knowledge, there were no similar cases reported in the literature. Case Presentation. A 65-year-old Tunisian man with no significant medical history consulted our department with a 10 cm ulcerative lesion on the base of the penis. The lesion biopsy was concordant with the diagnosis of basal cell carcinoma. The patient underwent a wide local excision. The tissue defect was reconstructed by an anterolateral pedicled flap of the thigh. The final histopathology report was concordant with the diagnosis of a trichoblastic carcinoma. Conclusion. There is no treatment consensus due to the few reported cases of trichoblastic carcinoma in the literature. Surgical excision with free margins remains the cornerstone of the treatment.
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