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Objective To investigate the clinicopathological features and clinical management of primary extragonadal germ cell tumor of the prostate. Methods Two cases of primary extragonadal germ cell tumor in the prostate were collected at Fudan University Shanghai Cancer Center, in January and September 2016, respectively. Their pathological features, clinical treatments and follow-up were retrospectively analyzed. Results The two patients were 41 and 32 years old, respectively, and both presented with obstructive symptoms of the lower urinary tract. Histol