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Malignant transformation of a mature cystic teratoma (MCT) of the ovary is extremely rare. Squamous cell carcinoma is the most common malignancy, which occurs in MCT and accounts for ∼80% of all cases. We describe the eighth undisputed case of a pure sebaceous carcinoma arising in a teratoma of the ovary. A 45-year-old woman underwent laparoscopic hysterectomy, bilateral salpingo-oophorectomy with pelvic and paraaortic lymphadenectomy. The final stage was pT1a, pN0 (0/30), M0, G2. The carcinoma was localized within a dermoid cyst not involving the capsule or adjacent abdominal structures. Complete tumor excision without iatrogenic tumor cell dissemination and proper staging are important for the prognosis and treatment and should be performed as soon as possible after diagnosis. Unfortunately, the very small number of cases published does not allow an evidence-based therapeutic management.