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Abstract  Ectopic ureter is a rare abnormality, so presenting a transitional cell carcinoma (TCC) arising from an ectopic ureter is extremely rare. We report here a case of a man with an invasive transitional cell carcinoma arising from a right ectopic ureter and managed by laparoscopy. To our knowledge, this is the fourth case described in the literature, and the second case of a TCC arising in a right ectopic ureter.