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Pure primary squamous cell carcinoma (SCCa) of the ovary not associated with pre-existing ovarian lesions including dermoid cysts, Brenner tumors, or endometriosis is extremely rare. The last 2 types of SCCa and pure primary SCCa have been classified by the World Health Organization criteria as surface epithelial-stromal tumors. On account of the rarity of pure primary SCCa of the ovary, the clinical features and effective treatments have not been established. This study describes 2 such cases that we recently encountered in a review of the literature. The literature shows that patients with advanced-stage tumors have very poor survival outcomes, despite a combination of surgery, radiation therapy, and chemotherapy.