Polyp Growing Inside the Lesion of Cystic Adenomyosis

A 44-year-old woman, gravida 3 para 1, presented to our hospital with a 20-year history of dysmenorrhea. Her surgical history revealed a cesarean section. Magnetic resonance imaging suggested a significantly enlarged uterus with cystic adenomyosis (Fig. 1). After complete assessment combined with informed consent, laparoscopic hysterectomy was performed. Subsequently, dissection of the surgical specimen demonstrated an irregularly shaped cystic space filled with brown fluid at the anterior lesion of cystic adenomyosis. An unexpected neoplasm with the size of 1.5 cm × 0.6 cm × 0.5 cm, which did not communicate with the uterine cavity, was visible inside the space (Fig. 2). The final pathological diagnosis of the specimens verified ectopic implantation of endometrial glands and stroma within uterine myometrium and a polyp formed on the ground of adenomyosis lesion (Fig. 3). Endometrial glands were considered partly cystic dilation in the case. The patient had an uneventful recovery and was discharged uneventfully 4 days after the surgery.Fig. 2Gross anatomy of the uterus. (A) A neoplasm (white arrow) was visible in lesion of cystic adenomyosis. (B) Uterine cavity.View Large Image Figure ViewerDownload Hi-res image Download (PPT)Fig. 3Histopathology of polyp growing on lesion of adenomyosis.View Large Image Figure ViewerDownload Hi-res image Download (PPT) The predilection site of polyp is the uterine cavity and cervix. Polyp found inside lesion of adenomyosis is a very rare pathological manifestation, which has never been reported before. Cystic adenomyosis is an unusual type of adenomyosis that presents as cystic growth in adenomyosis lesions, with endometrial glands in the walls of cysts [1Acién P Acién M Fernández F José Mayol M Aranda I The cavitated accessory uterine mass: a Müllerian anomaly in women with an otherwise normal uterus.Obstet Gynecol. 2010; 116: 1101-1109Google Scholar,2Zhao CZ Wang B Zhong CY Lu ST Lei L. Management of uterine cystic adenomyosis by laparoscopic surgery: case report.BMC Womens Health. 2021; 21: 263Google Scholar]. This special presentation makes us vigilant to attach importance to endometrial lesions inside cystic adenomyosis, such as endometrial polyps and carcinoma. Sporadic cases of cancer arising from adenomyosis have been previously reported [3Habiba M Pluchino N Petignat P Bianchi P Brosens IA Benagiano G. Adenomyosis and endometrial cancer: literature review.Gynecol Obstet Investig. 2018; 83: 313-328Google Scholar]. Dissection of the specimen and histopathological examination are essential for the excised tissues in patients undergoing surgery. If necessary, it is also suggested to perform fast frozen pathology to decrease misdiagnosis rate throughout surgery and determine scope of operation.