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Angioleiomyomas are common benign smooth muscle neoplasms in the dermis and subcutaneous tissue of limbs. Very rarely have they been described in the external male genitalia with only two cases in the paratesticular tissues and another intratesticular. We report a case of a paratesticular angioleiomyoma with cytological atypia and a peculiar vascular pattern, found incidentally as a painless nodule in a 75-year-old man. The main clinicopathological features together with the differential diagnoses and the pertinent published reports are reviewed.

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