The first case of penile leiomyosarcoma was described by Levi in 1930. In 1957 Ashley and Edwards reported the first case in the British literature and in 1963 Pack reported the first in the American literature. MacKenzie et al. were the first to recognize two types of leiomyosarcoma: superficial and deep. The first develop from smooth muscle cells from the superficial dermal layers of the glans penis or distal third of the penis, they are generally asymptomatic and less malignant, rarely invading deeper structures and without involvement of the urethra on physical exam. The best prognostic predictors are type of tumor at presentation (superficial vs. deep) and treatment choice, being total penectomy more effective for failures of local resections than for primary treatment of deep tumors.

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