Prophylactic oophorectomy and aromatase inhibitors for premenopausal deep angiomyxoma: A case report and literature review

Tonai,Nobutomo; Nasu,Kaei; Yano,Mitsutake; Sato,Miho; Kai,Kentaro; Nishida,Masakazu; Kawano,Yasushi

doi:10.3892/etm.2022.11702

Prophylactic oophorectomy and aromatase inhibitors for premenopausal deep angiomyxoma: A case report and literature review

Authors:
- Nobutomo Tonai
- Kaei Nasu
- Mitsutake Yano
- Miho Sato
- Kentaro Kai
- Masakazu Nishida
- Yasushi Kawano
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Affiliations: Department of Obstetrics and Gynecology, Oita University Faculty of Medicine, Yufu, Oita 879‑5593, Japan
Published online on: November 16, 2022 https://doi.org/10.3892/etm.2022.11702
Article Number: 3
Copyright: © Tonai et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

Deep angiomyxoma is a rare, infiltrative, hormone‑dependent, benign‑mesenchymal neoplasm that occurs in the deep soft tissues of the perineal regions. In total, 33% females with newly diagnosed deep angiomyxoma will typically relapse within 5 years after the standard treatment of radical resection. Postoperative hormone therapy is frequently administered to prevent recurrence, but the role of prophylactic oophorectomy in premenopausal women remain to be fully elucidated. In the present report, a 42‑year‑old Japanese woman was referred for a refractory Bartholin's cyst that is 14 cm in diameter. Based on the results of imaging (unenhanced CT and MRI) and histopathology, deep angiomyxoma was suspected, but no definitive diagnosis was possible. Tumor resection and bilateral salpingo‑oophorectomy were performed before the postoperative diagnosis was confirmed to be deep angiomyxoma. The patient received an aromatase inhibitor (2.5 mg letrozole daily) as adjuvant hormonal therapy. There was no evidence of recurrence at the 1‑year postoperative follow‑up. In conclusion, prophylactic oophorectomy and postoperative adjuvant therapy with aromatase inhibitors may be a promising treatment option for deep angiomyxoma to optimize the outcome of surgical treatment. Long‑term follow‑up is required to monitor for the late and/or local recurrence of deep angiomyxoma and possible adverse effects of adjuvant hormonal therapy.

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