Atypical Polypoid Adenomyoma in a Patient with Hyperprolactinemia: a Novel Case and Systematic Review of the Systematic Reviews.

Background: A 34-year-old nulligravid lady was referred to the outpatient gynecology department with the symptoms of abnormal menstrual bleeding and mild anemia. A transvaginal ultrasound (TVS) revealed the presence of a hyperechoic lesion measuring 9.5x4.5 mm in the uterine cavity at the level isthmus (lower uterine segment), suggestive of an endometrial polyp. Consequently, a diagnostic hysteroscopy was performed. Transcervical hysteroscopic resection (TCR) and diagnostic curettage (D&C) were accomplished. A macroscopic analysis of the specimen exhibited a white, pale polypoid formation measuring 1x1 cm with a hard consistency. Histology analysis confirmed the diagnosis of an atypical polypoid adenomyoma (APA). All these findings were explained to the patient and the risk of potential progression to endometrial carcinoma was discussed in detail. A multidisciplinary meeting (MDT) was held and conservative management with levonorgestrel coil insertion or Methoxy progesterone administration per os, along with frequent hysteroscopies every three months, was offered. The patient decided to undergo a hysterectomy with preservation of the ovaries, despite the lack of childbearing. A laparoscopic-assisted vaginal hysterectomy with preservation of adnexa was performed. The pathology analysis confirmed the diagnosis of APA with coexistence of well-differentiated (low-grade) endometrioid carcinoma of the endometrium, stage Ia. Methods: We performed a systematic review of systematic reviews with APA and quality assessment of the included studies. Furthermore, a flow-chart guideline for the management of APA in premenopausal and postmenopausal women is offered. Conclusions: Atypical polypoid adenomyoma is a rare clinical entity. It occurs mainly during the reproductive period of a woman. It is associated with abnormal vaginal bleeding, infertility, nulliparity, obesity, metabolic syndrome, hyperprolactinemia and hyperestrogenism. It is a risk factor for the development of endometrial hyperplasia and subsequent endometrial cancer. Preoperative diagnosis is extremely difficult; there are no specific imaging landmarks in ultrasound and magnetic resonance imaging (MRI) assessment. In patients with a desire for fertility, the management is complicated and should be based on a four-step hysteroscopic transcervical resection of the lesion. Hysterectomy is the right option for post-menopausal women and for women not wishing to conceive.