The incidence of subcutaneous leiomyosarcoma of the scrotum is very rare and they encompass a group of paratesticular leiomyosarcomas which account for only 2% of such cases. These tumors are categorized under subcutaneous superficial leiomyosarcomas and they usually do not involve the testis, epididymis or spermatic cord. Only 34 cases have been reported in literature worldwide till now, of which only 11 cases have been reported from India [1]. Considering its rarity, we had an unusual presentation of a 35-year-old male with a nodule over scrotum for 6months duration and was initially diagnosed as sebaeceous cyst on clinical examination. He then underwent excision for the same for which histopathological examination was done, which revealed the features of leiomyosarcoma and the same was confirmed by immunohistochemistry