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Introduction: Prostate cancer is the sixth most common cancer in the world, the second most common cancer among men, and the most common cancer in men in Europe. Metastatic prostate cancer among young patients represents the rarest of the newly diagnosed prostate cancer, with few reports of cases with a longer survival. Case presentation: We present the case of a 59 year-old male who was referred with back pain over the last month. Digital rectal examination highlighted an enlarged and totally indurated prostate of 4x4.5 cm, while abdominopelvic X-rays showed osteoblastic metastases in the spine and pelvis bones. Laboratory examinations revealed a Prostate Specific Antigen level of 7941 ng/ml. Prostate biopsy histology showed a bilateral prostate cancer with a Gleason score of 8. Androgen deprivation therapy and daily administration of biphosphonates were prescribed. After two years of treatment, the Prostate Specific Antigen level decreases to 8 ng/ml. Conclusions: We reported the highest Prostate Specific Antigen level in a patient under 60 years old with metastatic prostate cancer. Prostate cancer remains an important public health problem due to the aggressiveness of the disease and advanced stage upon diagnosis. Prostate Specific Antigen is mandatory to evaluate, to have a reference level in order to prevent metastatic prostate cancer in young patients at diagnosis.