Extrauterine adenomyoma: A case report and systematic review of the literature

ObjectiveTo describe a case of extrauterine adenomyoma (EA) and review all the cases of EA in the literature.MethodsPubmed/MEDLINE, Embase, and Google Scholar from 1807 to December 2022. All studies reporting the histologic diagnosis of an EA. We collected the following data: patient's age, size and location of adenomyoma, presence of endometriosis and adenomyosis, past gynecologic treatment, symptoms, diagnostic imaging, surgical intervention, alternative/adjuvant treatment, associated malignancy, and follow up.ResultsSixty-seven studies with 85 patients were included. Pain was the most frequent symptom (69.5%). Among diagnostic examinations, ultrasonography was used in 60 out of 81 reported cases, with several radiologic features described. EA was located inside the pelvis in 77.6% of patients. Adnexa were the most frequent site of the disease (24, 28.2%). History of endometriosis or adenomyosis was described in 35 patients (35, 41.2%). Uterine tissue morcellation was reported in 6 of the 85 patients (7.1%). Associated malignancy was detected in 9 out of 85 patients with available data (10.6%). There were two recurrences of disease.ConclusionSpecific imaging features of EA are yet to be described in the literature. History of endometriosis and adenomyosis or uterine tissue morcellation may be suggestive of EA. Histologic examination can give a definitive diagnosis and exclude malignant transformation. Extrauterine adenomyoma is a rare disease with a challenging preoperative diagnosis due to its non-specific clinical characteristics.