A case of male perineal aggressive angiomyxoma with expressions of female hormone receptors

Abstract

Introduction Aggressive angiomyxoma is a rare mesenchymal tumor in females of reproductive age that occurs in the pelvis and perineal zone with a high risk of local infiltration and recurrence. Male aggressive angiomyxoma in perineal zone is very rare. Case presentation A 63‐year old male presented to our hospital with chief complaint of perineal mass. He was diagnosed with perineal lipoma by needle biopsy 3 years before. Computed tomography revealed a 16 cm perineal tumor without lymph node or distal metastasis. As the tumor had gradually increased, we performed tumor resection. The histological diagnosis of tumor using immunohistochemistry was aggressive angiomyxoma with complete resection. Moreover, expressions of both estrogen and progesterone receptors were recognized. No recurrence was seen 9 months after surgery. Conclusion Male aggressive angiomyxoma is a very rare tumor, which has expressions of female hormone receptors. Hormonal therapy might be effective for perioperative therapy or recurrence.

Characteristics and outcomes of patients with primary abdominopelvic aggressive angiomyxoma: a retrospective review of 12 consecutive cases from a sarcoma referral center

“…No case developed with distant metastasis. Besides, AAM is a likely hormone dependent tumor [ 21 , 22 ]. Estrogen receptor (ER) and progesterone receptor (ER) positive were confirmed in 91.7% (11/12) of patients (including males).…”