• Antonio Raffone, Antonio Travaglino, Gabriele Saccone, Carlo Alviggi, Massimo Mascolo, Giuseppe De Placido, Luigi Insabato, Antonio Mollo, and Fulvio Zullo.
• Gynecology and Obstetrics Unit, Department of Neuroscience, Reproductive Sciences and Dentistry, School of Medicine, University of Naples Federico II, Naples, Italy.
• Acta Obstet Gynecol Scand. 2019 Jul 1; 98 (7): 842-855.

IntroductionAtypical polypoid adenomyoma is an uncommon uterine lesion which can coexist with endometrial atypical hyperplasia and/or cancer. Atypical polypoid adenomyoma affects premenopausal women in most cases, but it shows high recurrence rate if conservatively treated. To date, the management of patients is based on low-quality evidence and is not standardized. Our primary aim was to explore the optimal management of atypical polypoid adenomyoma, with particular regard to the fertility-sparing approach. The secondary aim was to define clinicopathologic features of atypical polypoid adenomyoma.Material And MethodsMedline, Embase, Web of Sciences, Scopus, ClinicalTrial.gov, OVID, Google Scholar and Cochrane Library were searched for studies reporting outcomes of atypical polypoid adenomyoma treatments. Univariate comparisons among outcomes of fertility-sparing treatments (rates of initial response, progression, recurrence, final complete response, pregnancy) were performed with Fisher's exact test (α = .05).ResultsEleven retrospective studies with 237 patients were included; 85.5% of patients were premenopausal and 62.9% were nulliparous. Atypical polypoid adenomyoma coexisted with atypical hyperplasia in 5.5% of cases and with endometrial cancer in 5.9%. Overall risks of recurrence and progression to cancer were 28.9% and 16.6%, respectively. Fertility-sparing treatments included hormonal therapy with or without maintenance, hysteroscopic transcervical resection, dilation and curettage, and hormonal therapy combined with transcervical resection or dilation and curettage. Transcervical resection showed significantly higher initial response rates (P from <0.001 to 0.023) than any other treatment. Transcervical resection and transcervical resection+hormonal therapy showed significantly lower progression rates (P < 0.001), and higher final complete response rates (P < 0.001) than any other treatment. No significant differences were found in the rates of pregnancy (P = 0.533 - 0.647) or recurrence (P = 0.052 - 0.475). Among the different transcervical resection techniques, the 4-step transcervical resection showed significantly lower rates of progression (P = 0.002) and recurrence (P = 0.013) than other techniques. Limitations to our results were the retrospective design of the studies and the relatively small sample size, due to the rarity of atypical polypoid adenomyoma.ConclusionsBased on its effectiveness and safety, transcervical resection may be the first-line fertility-sparing treatment for atypical polypoid adenomyoma. In particular, 4-step transcervical resection showed the best results. Given the risk of recurrence, progression and coexistent atypical hyperplasia or cancer, follow-up biopsies are advisable. When fertility preservation is not required, hysterectomy might be advisable.© 2019 Nordic Federation of Societies of Obstetrics and Gynecology.

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