Aggressive angiomyxoma is a rare benign mesenchymal stromal tumour, characterized by locally infiltrative nature and a tendency for recurrence. Only a few cases of penile involvement have been reported in the literature so far. We report a case of aggressive penile angiomyxoma in a sixty-two-year-old obese, diabetic male patient. He presented with obstructive lower urinary tract symptoms (LUTS) and diffuse enlargement of the penis and scrotum. He was managed with excision, reduction scrotoplasty, internal urethrotomy, followed by Leuprolide therapy for prevention of recurrence. He is on follow up for 20 months without recurrence and obstructive symptoms.
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