Aggressive angiomyxoma (AAM) is a rare mesenchymal benign myxoid tumor, characterized by locally infiltrative nature and high recurrence rate. AAM occurs almost exclusively in adult females, arising from the soft tissue of the pelvic region: to our knowledge, only 43 cases occurring in men have been reported. We report a case of massive recurrence of scrotal AAM in a 46-year-old obese man, who already underwent surgery for the same disease in 2004 and 2005. The mass had a circumference of 106 cm and weighted 30 kg. It was impossible to appreciate the testes and to find the penis. The patient underwent scrotal resection, bilateral orchidopexia and transposition of the penis, by means of a preputial flap. Residual scrotal skin was modeled in order to create a neoscrotum, where the testes were placed and secured with interrupted sutures. Histologic examination showed diffuse angiomyxoma-like lipomatosis. After three months, the patient presented with local relapse which also involved the external urethral orifice.
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