Introduction A glioblastoma arising from an ovarian mature cystic teratoma is extremely rare. Guidelines for treatment of glioblastoma arising in teratoma have not been established because of its rarity. We report a case of glioblastoma arising from an ovarian teratoma diagnosed at Stage IA and we review the literature on glioblastoma arising from an ovary. Patients A 31-year-old woman (gravida 3, para 1) visited a hospital because of abdominal fullness. She had no significant medical or gynecological history. Magnetic resonance imaging revealed the bilateral ovary tumor suggested mature cystic teratoma. She underwent total hysterectomy with bilateral salpingo-oophorectomy and partial omentectomy because frozen section diagnosis during surgery was immature teratoma of the left ovary. Based on the morphology and immunohistochemical results, the final diagnosis was a glioblastoma arising from an ovarian teratoma. She was recommended additional treatment options, but she chose close observation. Six months later, she relapse, which was diagnosed by Computed tomography imaging of an enlarged para-aortic lymph node. She underwent systematic para-aortic and pelvic lymphadenectomy. The metastatic lymph node showed pathological features similar to those of the primary tumor. She was administered temozolomide (TMZ) for maintenance therapy followed by TMZ with concomitant radiotherapy. After 6 months, CT indicated ascites and lymph node metastasis. She received a carboplatin+etoposide regimen. Results After 3 cycles of a carboplatin+etoposide regimen, her diseases were remarkably reduced. Conclusion To our knowledge, this is only the fifteenth reported case of glioblastoma occurring in an ovarian mature cystic teratoma. There is no consensus regarding the most appropriate treatment for recurrent glioblastoma arising from the ovary. The regimen of carboplatin+etoposide may be an alternative choice for patients with recurrent high-grade glioma.
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