Background: Prostate cancer is the second most common cause of cancer death in men, being responsible for approximately 13% of all cancer deaths. Due to the high specificity for prostate tissue, prostate-specific antigen (PSA) is the primary serum tumor marker for prostate cancer. To our knowledge, in published data, the highest reported PSA level on initial presentation was 5,666 ng/ml. Case Report: We present a 64-year-old Caucasian man with no specific urologic or pain symptoms and with an initial PSA serum value of 21,380 ng/ml. Initial laboratory studies showed chronic anemia and elevated alkaline phosphatase, most likely from bone marrow infiltration due to metastatic disease. Prostate biopsies diagnosed an adenocarcinoma of the prostate (Gleason score 9). Computed tomography (CT) showed multiple liver metastases with retroperitoneal lymph nodes of up to 1 cm. The prostate was moderately enlarged (estimated weight 35 g). Both kidneys were normal (no hydronephrosis present). A bone scintigraphy demonstrated diffuse osseous metastasis. Treatment was initiated with bilateral subcapsular orchiectomy and bicalutamide therapy in an effort to create total androgen blockade. After 3 months of follow-up, the serum concentration of PSA was 29 ng/ml. Conclusion: This case presents an asymptomatic prostate cancer patient with bone and liver metastasis, enlarged retroperitoneal lymph nodes and the highest PSA level published to date.

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