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Skeletal muscle metastasis of prostate cancer is a very rare phenomenon that has only been described in limited case reports. In this study, we present a case of neuroendocrine prostate cancer with muscle metastasis, a histological subtype associated with a grim prognosis. This case illustrates the potential efficacy of urgent surgical resection of the metastatic muscle mass, followed by adjuvant radiation therapy, as a suitable management strategy for this condition. However, a comprehensive understanding of the biological characteristics of neuroendocrine prostate cancer is imperative in our fight against this lethal form of the disease and in the prevention of metastatic spread.