Objective: To report a case of a neuroendocrine differentiation in a prostate cancer patient, a rare subtype.

Methods: We describe the case of a patient diagnosed with adenocarcinoma of the prostate initially, who presented hematuria due to disease progression with neuroendocrine differentiation despite androgen-deprivation therapy (ADT ).

Discussion: Prostate cancer is the most common tumor in men. Histologically they are diagnosed as adenocarcinomas, which followed by ADT for a long time, develop neuroendocrine differentiation (NED ).

Conclusions: The prognostic significance of NED remains controversial. We must think in neuroendocrine differentiation in ADT-treated patient with disease progression and low PSA.

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