We report a case of a urothelial carcinoma with trophoblastic differentiation of the bladder that occured in a 23-year-old man. The patient presented with hematuria. Multiple papillary masses were resected transurethrally. Histopathologically, grade III urothelial cell carcinoma contained giant cells that were positive for human chorionic gonadotrophin (HCG), placental alkalen phosphatase, and human placental lactogen. HCG secreting tumors are reported to be highly aggressive. The patient is alive and well without evidence of recurrent disease or metastasis at 10 months from transurethral resection. To our knowledge, this case is the youngest patient in the literature so far.

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