Multifocal Polypoid Endometriosis Mimicking Malignancy in a Young Woman with a History of Hormonal Treatment

Polypoid endometriosis is a rare type of endometriosis. In con- trast to the usual cystic endometriosis, it is solid and frequently forms a nodular polypoid mass that can sometimes mimic ma- lignancy at presentation.1 Frequently involved sites are the vagi- Corresponding Author Jeong Yun Shim, MD, PhD Department of Pathology, CHA Gangnam Medical Center, 566 Nonhyeon-ro, Gangnam-gu, Seoul 06135, Korea Tel: +82-2-3468-2612, Fax: +82-2-3468-2619, E-mail: [email protected] Received: March 19, 2015 Revised: April 30, 2015 Accepted: May 12, 2015 http://jpatholtm.org/http://dx.doi.org/10.4132/jptm.2015.05.12 Polypoid Endometriosis Mimicking Malignancy • 419 na, cervix, adnexa, and colorectum.2-4 It usually causes non-spe- cific symptoms, such as dysmenorrhea, menorrhagia, or vaginal spotting.1,3,5 The etiology of polypoid endometriosis is uncertain. Some re- ported cases have been associated with unopposed estrogen treat- ment, phytoestrogen, and tamoxifen, suggesting the role of es- trogenic stimulation in the development of the disease.1,3-6 With- drawal of a GnRH agonist was also reported to cause polypoid endometriosis as a rebound phenomenon.3,7 To our knowledge, there is no previous report of polypoid endometriosis associated with OCs. In a collective review of 22 cases of polypoid endo- metriosis, Parker et al.1 included four cases with mixed estrogen- progestin therapy. But it was in the form of hormone replace- ment therapy after menopause or oophorectomy rather than a contraceptive use. OCs, which are frequently used to prevent the recurrence of endometriosis after conservative surgery, have also been reported to increase the risk of endometriosis in certain conditions.8 In our patient, estrogen seems to have retained its Fig. 1. (A) On axial computed tomography, a solid and cystic mass (arrows) with periuterine adhesions is visible in the left adnexa. (B) Anoth- er mass can be seen in the left upper abdominal wall (arrows) and extending to the rectus abdominis muscle. A small amount of ascites is present in the perihepatic area (arrowheads). (C) Microscopically, the mass is composed of endometrial-like tissue. Dilated endometrial-type glands are longitudinally arranged around grouped, thick-walled vessels with swollen and congested stroma, which is reminiscent of a typi- cal endometrial polyp. (D) Some glandular epithelial cells demonstrate ciliated metaplasia. (E) On immunohistochemical staining, the glandu- lar epithelial cells and some stromal cells are positive for estrogen receptor. (F) CD10 is positive only in stromal cells. A C E B D F http://jpatholtm.org/ http://dx.doi.org/10.4132/jptm.2015.05.12 420 • Kim J-Y, et al. stimulatory effect despite being balanced with progesterone. Polypoid endometriosis can sometimes mimic the clinical pre- sentation of malignancy.1 The presence of a large adnexal mass, multiple peritoneal and pelvic foci, ascites, and elevated CA 125 in our case gave the preoperative impression of a malignant ovar- ian tumor with peritoneal metastases. An elevated CA 125 level is one of the laboratory abnormalities associated with endome- triosis and is also reported in some cases of polypoid endometri- osis.4,9 It has been reported that ascites, though very rarely, can be associated with endometriosis, especially in the presence of severe disease.10 When it presents with ascites, particularly ac- companied by multiple peritoneal lesions on radiology, adnexal polypoid endometriosis can be easily misinterpreted as ovarian malignancy with peritoneal metastases. Our patient underwent laparoscopic surgery despite the pre- operative suspicion of malignancy. Gynecological laparoscopy is increasingly performed, even in some early oncological condi- tions, because of its reduced invasiveness. In our case, an intraop- erative frozen diagnosis of a benign mass prevented conversion of the procedure to a more extensive surgery with laparotomy. De- spite the relatively typical microscopic findings, the confusing presentation of the mass, such as its large size, solid nodular na- ture, destruction of normal anatomical structures, multifocality, peritoneal involvement, ascites, and elevated CA 125, rendered doubt on the benign nature of the lesion. Pathologists’ awareness of this rare form of endometriosis is vital in order to avoid un- necessary radical surgery. Yet again, a thorough microscopic ex- amination with sufficient sampling is warranted in these patients in order to exclude complex hyperplasia or adenocarcinoma, which, although very rare, has arisen in polypoid endometrio- sis.1 Polypoid endometriosis should also be included in the dif- ferential diagnosis of seemingly malignant adnexal tumors in a young woman, especially when associated with a history of hor- mone therapy, notably OCs, for endometriosis. Conflicts of Interest No potential conflict of interest relevant to this article was reported.

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