{"paper_id":"a2536a4d-f836-42b6-88a5-b489a407f619","body_text":"418\npISSN 2383-7837\neISSN 2383-7845\n© 2015 The Korean Society of Pathologists/The Korean Society for Cytopathology\nThis is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/ \nby-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.\nMultifocal Polypoid Endometriosis Mimicking Malignancy in a Young \nWoman with a History of Hormonal Treatment\nJi-Young Kim · Tae-Jong Song1 · Hye-Kyung Choi2 · Jeong Yun Shim\nDepartment of Pathology, CHA Gangnam Medical Center, CHA University School of Medicine, Seoul;  \n1Department of Obstetrics and Gynecology, Kangbuk Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul;  \n2Department of Radiology, CHA Gangnam Medical Center, CHA University School of Medicine, Seoul, Korea\nJournal of Pathology and Translational Medicine 2015; 49: 418-420\nhttp://dx.doi.org/10.4132/jptm.2015.05.12\n▒ BRIEF CASE REPORT ▒\nPolypoid endometriosis is a rare type of endometriosis that \nsometimes mimics the clinical presentation of malignancy. We \nreport a case of a young woman with a history of hormone treat-\nment who was suspected to have an ovarian malignancy with \nmetastases.\nCASE REPORT\nA 30-year-old G0P0 woman with a history of left ovarian \ncystectomy due to endometriosis presented with a huge left ad-\nnexal mass detected on ultrasonography. After the previous cys-\ntectomy, she was administered a gonadotrophin releasing hor-\nmne (GnRH) agonist for four months and then a combined oral \ncontraceptive (OC), Mercilon (Organon Ltd., Doublin, Ireland) \n(mixed ethinylestradiol and desogestrel), for about two years. \nHer menstrual cycle was regular, but she experienced severe \ndysmenorrhea. The patient’s serum cancer antigen (CA) 125 level \nwas increased (595.3 U/mL), while her CA 19-9 and carcinoem-\nbryonic antigen results were normal.\nUltrasonography showed an 11-cm-sized, mixed-echoic, semi-\nsolid mass in the left adnexa. Abdominopelvic computed to-\nmography revealed the mass to be solid with enhancement (Fig. \n1A). Another mass was present in the left abdominal wall, ex-\ntending to the peritoneum and the rectus abdominis muscle. A \nsmall amount of ascites was observed in the perihepatic space \n(Fig. 1B).\nA laparoscopic left salpingo-oophorectomy was performed to \nremove the 11-cm-sized tumor encasing and destroying the ad-\nnexal organs. Intraoperative frozen section examination indicat-\ned the lesion to be benign, most likely severe endometriosis. \nOther peritoneal foci were also resected.\nMicroscopically, the left adnexal mass was composed of endo-\nmetrial-like tissue. Dilated endometrial-type glands were lon-\ngitudinally arranged around grouped, thick-walled vessels, which \nwere reminiscent of a typical endometrial polyp (Fig. 1C). Wor-\nrisome features, such as increased stromal cellularity or atypia, \nwere not noted. The mitotic rate was less than 1 in 10 high-\npower fields (Fig. 1D). Immunohistochemically, both the epi-\nthelial and the stromal cells were positive for estrogen receptor \nand progesterone receptor. The stromal cells were also positive \nfor CD10 (Fig. 1E, F). The absence of increased cellularity and/\nor stromal atypia easily excluded the possibility of adenosarcoma. \nThe endometrial-type stromal cells with CD10 positivity in our \ncase were obviously different from the fibroblastic and CD34-\npositive stromal cells in adenofibroma.\nThe patient did not present residual disease at her follow-up \nsix months after the operation.\n \nDISCUSSION\nPolypoid endometriosis is a rare type of endometriosis. In con-\ntrast to the usual cystic endometriosis, it is solid and frequently \nforms a nodular polypoid mass that can sometimes mimic ma-\nlignancy at presentation.1 Frequently involved sites are the vagi-\nCorresponding Author\nJeong Yun Shim, MD, PhD\nDepartment of Pathology, CHA Gangnam Medical Center, 566 Nonhyeon-ro, \nGangnam-gu, Seoul 06135, Korea \nTel: +82-2-3468-2612, Fax: +82-2-3468-2619, E-mail: jyshim@cha.ac.kr\nReceived: March 19, 2015  Revised: April 30, 2015\nAccepted: May 12, 2015\n\n\nhttp://jpatholtm.org/http://dx.doi.org/10.4132/jptm.2015.05.12\nPolypoid Endometriosis Mimicking Malignancy  •     419\nna, cervix, adnexa, and colorectum.2-4 It usually causes non-spe-\ncific symptoms, such as dysmenorrhea, menorrhagia, or vaginal \nspotting.1,3,5\nThe etiology of polypoid endometriosis is uncertain. Some re-\nported cases have been associated with unopposed estrogen treat-\nment, phytoestrogen, and tamoxifen, suggesting the role of es-\ntrogenic stimulation in the development of the disease.1,3-6 With-\ndrawal of a GnRH agonist was also reported to cause polypoid \nendometriosis as a rebound phenomenon.3,7 To our knowledge, \nthere is no previous report of polypoid endometriosis associated \nwith OCs. In a collective review of 22 cases of polypoid endo-\nmetriosis, Parker et al.1 included four cases with mixed estrogen-\nprogestin therapy. But it was in the form of hormone replace-\nment therapy after menopause or oophorectomy rather than a \ncontraceptive use. OCs, which are frequently used to prevent the \nrecurrence of endometriosis after conservative surgery, have also \nbeen reported to increase the risk of endometriosis in certain \nconditions.8 In our patient, estrogen seems to have retained its \nFig. 1. (A) On axial computed tomography, a solid and cystic mass (arrows) with periuterine adhesions is visible in the left adnexa. (B) Anoth-\ner mass can be seen in the left upper abdominal wall (arrows) and extending to the rectus abdominis muscle. A small amount of ascites is \npresent in the perihepatic area (arrowheads). (C) Microscopically, the mass is composed of endometrial-like tissue. Dilated endometrial-type \nglands are longitudinally arranged around grouped, thick-walled vessels with swollen and congested stroma, which is reminiscent of a typi-\ncal endometrial polyp. (D) Some glandular epithelial cells demonstrate ciliated metaplasia. (E) On immunohistochemical staining, the glandu-\nlar epithelial cells and some stromal cells are positive for estrogen receptor. (F) CD10 is positive only in stromal cells.\nA\nC\nE\nB\nD\nF\n\nhttp://jpatholtm.org/ http://dx.doi.org/10.4132/jptm.2015.05.12\n420     •  Kim J-Y, et al.\nstimulatory effect despite being balanced with progesterone.\nPolypoid endometriosis can sometimes mimic the clinical pre-\nsentation of malignancy.1 The presence of a large adnexal mass, \nmultiple peritoneal and pelvic foci, ascites, and elevated CA 125 \nin our case gave the preoperative impression of a malignant ovar-\nian tumor with peritoneal metastases. An elevated CA 125 level \nis one of the laboratory abnormalities associated with endome-\ntriosis and is also reported in some cases of polypoid endometri-\nosis.4,9 It has been reported that ascites, though very rarely, can \nbe associated with endometriosis, especially in the presence of \nsevere disease.10 When it presents with ascites, particularly ac-\ncompanied by multiple peritoneal lesions on radiology, adnexal \npolypoid endometriosis can be easily misinterpreted as ovarian \nmalignancy with peritoneal metastases.\nOur patient underwent laparoscopic surgery despite the pre-\noperative suspicion of malignancy. Gynecological laparoscopy is \nincreasingly performed, even in some early oncological condi-\ntions, because of its reduced invasiveness. In our case, an intraop-\nerative frozen diagnosis of a benign mass prevented conversion of \nthe procedure to a more extensive surgery with laparotomy. De-\nspite the relatively typical microscopic findings, the confusing \npresentation of the mass, such as its large size, solid nodular na-\nture, destruction of normal anatomical structures, multifocality, \nperitoneal involvement, ascites, and elevated CA 125, rendered \ndoubt on the benign nature of the lesion. Pathologists’ awareness \nof this rare form of endometriosis is vital in order to avoid un-\nnecessary radical surgery. Yet again, a thorough microscopic ex-\namination with sufficient sampling is warranted in these patients \nin order to exclude complex hyperplasia or adenocarcinoma, \nwhich, although very rare, has arisen in polypoid endometrio-\nsis.1 Polypoid endometriosis should also be included in the dif-\nferential diagnosis of seemingly malignant adnexal tumors in a \nyoung woman, especially when associated with a history of hor-\nmone therapy, notably OCs, for endometriosis.\nConflicts of Interest\nNo potential conflict of interest relevant to this article was \nreported.\nREFERENCES\n1. Parker RL, Dadmanesh F, Young RH, Clement PB. Polypoid endo-\nmetriosis: a clinicopathologic analysis of 24 cases and a review of \nthe literature. Am J Surg Pathol 2004; 28: 285-97.\n2. Jaiman S, Gundabattula SR, Pochiraju M, Sangireddy JR. Polypoid \nendometriosis of the cervix: a case report and review of the litera-\nture. Arch Gynecol Obstet 2014; 289: 915-20.\n3. Othman NH, Othman MS, Ismail AN, Mohammad NZ, Ismail Z. \nMultiple polypoid endometriosis: a rare complication following \nwithdrawal of gonadotrophin releasing hormone (GnRH) agonist \nfor severe endometriosis: a case report. Aust N Z J Obstet Gynaecol \n1996; 36: 216-8.\n4. Kraft JK, Hughes T. Polypoid endometriosis and other benign gyn-\naecological complications associated with tamoxifen therapy: a \ncase to illustrate features on magnetic resonance imaging. Clin Ra-\ndiol 2006; 61: 198-201.\n5. Felix A, Nogales FF, Arias-Stella J. Polypoid endometriosis of the \nuterine cervix with Arias-Stella reaction in a patient taking phy-\ntoestrogens. Int J Gynecol Pathol 2010; 29: 185-8.\n6. Chang CK, Chen P , Leu FJ, Lou SM. Florid polypoid endometriosis \nexacerbated by tamoxifen therapy in breast cancer. Obstet Gynecol \n2003; 102(5 Pt 2): 1127-30.\n7. Marugami N, Hirohashi S, Kitano S, et al. Polypoid endometriosis \nof the ureter mimicking fibroepithelial polyps. Radiat Med 2008; \n26: 42-5.\n8. Tu FF, Du H, Goldstein GP , Beaumont JL, Zhou Y, Brown WJ. The \ninfluence of prior oral contraceptive use on risk of endometriosis is \nconditional on parity. Fertil Steril 2014; 101: 1697-704.\n9. Laird LA, Hoffman JS, Omrani A. Multifocal polypoid endometri-\nosis presenting as huge pelvic masses causing deep vein thrombo-\nsis. Arch Pathol Lab Med 2004; 128: 561-4.\n10. Gungor T, Kanat-Pektas M, Ozat M, Zayifoglu Karaca M. A sys-\ntematic review: endometriosis presenting with ascites. Arch Gyne-\ncol Obstet 2011; 283: 513-8.","source_license":"CC0","license_restricted":false}