Multiple extrauterine adenomyomas and uterus-like masses: case reports and review of the literature.

OBJECTIVE

To describe two rare cases of multiple extrauterine adenomyomas, their clinical management, and their response to surgical and hormone therapy with GnRH agonist.

DESIGN

Case report and review of the English-language scientific literature.

SETTING

Cases presented in a tertiary health care academic institution.

PATIENT(S): Two patients without urogenital malformations diagnosed with extrauterine adenomyoma, which is a benign tumor composed of smooth muscle and endometrium, typically originating within the uterus.

INTERVENTION(S): Surgical radical excision of adenomyomas followed by a long-term hormone therapy with GnRH agonist.

MAIN OUTCOME MEASURE(S): Anatomical and clinical outcomes.

RESULT(S): Surgical treatment followed by long-term GnRH agonist therapy appeared effective in keeping the disease stable. After a long-term follow-up of 10 and 4 years, the two patients are still asymptomatic and stable.

CONCLUSION(S): Only 19 other cases of extrauterine adenomyomas are reported in the English-language scientific literature, and no cases of multiple masses are described until now. It is probable that these extrauterine adenomyomas arose from a metaplastic transformation of the subcelomic mesenchyme. Long-term GnRH agonist therapy after surgery appeared effective in keeping the disease stable.