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This study investigates a unique case of penile sarcoma, providing a comprehensive morphological and molecular analysis. Through the creation of experimental patient-derived models\u2014including patient-derived xenograft (PDX), 3D, and monolayer primary cultures\u2014we successfully replicated crucial molecular traits observed in the patient\u2019s tumor, such as smooth muscle actin and CD99 expression, along with specific mutations in genes like TSC2 and FGFR4. These models are helpful in assessing the potential for an in-depth exploration of this tumor\u2019s biology. 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