{"paper_id":"fb9c0ca7-db40-4864-8d09-88016ae0f43d","body_text":"Articles published in Obstet Gynecol Sci are open-access, distributed under the terms of \nthe Creative Commons Attribution Non-Commercial License (http://creativecommons.\norg/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, \nand reproduction in any medium, provided the original work is properly cited.\nCopyright © 2015 Korean Society of Obstetrics and Gynecology \nCase Report\nObstet Gynecol Sci 2015;58(4):327-330\nhttp://dx.doi.org/10.5468/ogs.2015.58.4.327\npISSN 2287-8572 · eISSN 2287-8580\nwww.ogscience.org 327\nIntroduction\nTamoxifen has been widely used for adjuvant treatment of \nbreast cancer, but several gynecological side effects after its \nuse have been noted, including endometrial hyperplasia, endo-\nmetrial polyps, endometrial carcinoma, and rare complications \nsuch as endometriosis, adenomyosis, and cervical polyps [1-3]. \nPolypoid endometriosis is a rare benign gynecological disease, \nand several reported cases were associated with the use of \ntamoxifen [2]. To the best of our knowledge, three cases of \npolypoid endometriosis have been reported in the Korean lit-\nerature, but in none of the cases, polypoid endometriosis was \nassociated tamoxifen use. Herein, we report the first case of \npolypoid endometriosis associated with tamoxifen treatment in \na breast cancer patient.\nCase report\nA 66-year-old woman, who had received left partial mastectomy \ndue to ductal carcinoma in situ about 4 years ago and was tak-\ning tamoxifen treatment until a recent date, was admitted for \nevaluation of an incidentally detected ovarian cyst. She had \nreached menopause 10 years ago and had no other medical \nhistory except that she was a carrier of hepatitis C virus. In the \nlaboratory findings, liver enzymes showed an occasional increase \nbut there was no specific problem, and CA-15-3 was within the \nnormal range. Although the initial pelvic sonography in breast \ncancer work-up showed no abnormal findings, the second pelvic \nsonography performed 2 months before hysterectomy revealed \nthickened endometrium (9.7 mm), suggesting endometrial hy-\nperplasia, and a right ovarian cyst (41×36×35 mm). Pelvic mag-\nnetic resonance imaging revealed multiple cystic structures in \nthe endometrial cavity with mildly thickened endometrium and \nan approximately 4.1×3.4-cm-sized, well-defined, multicystic le-\nReceived: 2014.10.2.   Revised: 2015.1.15.   Accepted: 2015.2.6.\nCorresponding author: Dong Won Kim\nDepartment of Pathology, Soonchunhyang University Seoul \nHospital, 59 Daesagwan-ro, Y ongsan-gu, Seoul 140-743, Korea\nTel: +82-2-709-9426   Fax: +82-2-709-9441\nE-mail: budapast@schmc.ac.kr\nTamoxifen-associated polypoid endometriosis mimicking \nan ovarian neoplasm\nIn Ho Choi\n1\n, So-Young Jin\n1\n, Yoon Mi Jeen\n1\n, Jeong Jae Lee\n2\n, Dong Won Kim\n1\nDepartments of \n1\nPathology, \n2\nObstetrics and Gynecology, Soonchunhyang University Seoul Hospital, Soonchunhyang University College of Medicine, Seoul, \nKorea\nTamoxifen has been widely used for adjuvant treatment of breast cancer, but several gynecological side effects have \nbeen noted, including endometrial hyperplasia, polyp and carcinoma. Polypoid endometriosis is one of the extremely \nrare benign complications associated with tamoxifen therapy. A 66-year-old postmenopausal woman, who had received \nleft partial mastectomy due to breast cancer (about 4 years ago) and was taking tamoxifen treatment, had an ovarian \ncyst on ultrasonography. Pelvic magnetic resonance imaging suggested tamoxifen-associated endometrial and ovarian \nchanges, especially a 4.1×3.4-cm-sized, well-defined, multicystic mass in the right ovary. She received hysterectomy with \nbilateral salpingo-oophorectomy. Microscopically, the right paratubal mass showed endometrial glands and stroma, \nand immunohistochemical staining for CD10 confirmed the endometrial nature of the stroma. Three cases of polypoid \nendometriosis have been reported in the Korean literature, but in none of the cases, polypoid endometriosis was \nassociated with tamoxifen use. Herein, we report the first case of polypoid endometriosis associated with tamoxifen \ntreatment in Korea.\nKeywords: Endometriosis; Polyps; Tamoxifen\n\nwww.ogscience.org328\nVol. 58, No. 4, 2015\nsion in the right ovary with peripheral enhancement, suggestive \nof tamoxifen-associated endometrial and ovarian changes (Fig. \n1). Under the impression of endometrial hyperplasia and right \novarian cyst, she received hysterectomy with bilateral salpingo-\noophorectomy. The right ovarian mass, which was detected by \nradiologic examination, was confirmed to be a right paratubal \nmass.\nGrossly, a 4×3-cm-sized, well-circumscribed, polypoid mass \nwas noted in the right paratubal area (Fig. 2A). Microscopically, \nit revealed a multilocular cystic structure comprising of cystically \ndilated endometrial glands and stroma together (Fig. 2B). No hy-\nperplastic feature or complexity of glands was noted. There were \nno areas of fibrosis, necrosis, and decidual change in the stroma. \nThe glands were lined by a single layer of columnar or cuboidal \ncells with no identifiable cellular atypia or mitosis (Fig. 2C). Both \nglandular epithelial cells and stromal cells were positive for estro-\ngen receptor (not shown in figure). Presence of CD10-positive \nstromal cells confirmed the endometrial nature (Fig. 2D). She was \ndiagnosed as having an endometrial polyp with cervical endome-\ntriosis and paratubal polypoid endometriosis and was discharged \nwithout any complications.\nDiscussion\nEndometriosis is an estrogen-dependent disorder, and it is \nrare in postmenopausal women. There are a growing number \nof cases in the literature, which suggest that the tamoxifen \nis an antagonist of the estrogen receptor in breast tissue but \nbehaves as an agonist in the endometrium, and is associated \nwith the development of endometriosis in post-menopausal \nwoman with breast cancer [3-5].\nSince Mostoufizadeh and Scully [6] first described polypoid \nendometriosis in 1980, it has been considered as a benign \ngynecologic lesion that develops in endometriosis. It usually \nsimulates a malignant tumor during the operation and his -\ntologically resembles an endometrial polyp. According to the \nreview of 24 cases of polypoid endometriosis by Parker et al. [7], \npolypoid endometriosis showed several clinico-pathologic dif-\nferences when compared with typical endometriosis. It usually \noccurred in an older age group; 60% of patients with polypoid \nendometriosis in their study were more than 50 years of age \nas compared to <5% of patients with typical endometriosis. \nThe authors also mentioned that hormonal factors might play \na role in its pathogenesis, including its association with tamoxi-\nfen treatment.\nPathologically, polypoid endometriosis should be differentiated \nfrom other neoplasms, such as adenofibroma and adenosarcoma \ndue to the presence of benign glands. Indeed, focal or diffuse \narchitectural atypia of the glandular epithelium can be observed \nin the above lesions. However, endometrioid adenofibroma usu-\nally has an ovarian-type stroma in contrast to the endometrial-\ntype stroma of polypoid endometriosis. Adenosarcoma can be \ndistinguished from polypoid endometriosis because the former \nhas projecting stromal papillae or broad polypoid fronds, atypia \nof stromal cells, and periglandular cellular stromal cuffs.\nFig. 1. (A,B) Pelvic dynamic magnetic resonance imaging shows multiple subendometrial cystic changes with mildly thickened endometrium and a \n4.1×3.4-cm-sized, well-defined, multicystic lesion in the right adnexal portion.\nA B\n\nwww.ogscience.org 329\nIn Ho Choi, et al. Tamoxifen treatment-associated polypoid endometriosis \nIn conclusion, three cases of polypoid endometriosis have been \nreported in Korea; in two cases, polypoid endometriosis origi-\nnated from the rectum mimicking rectal cancer, and in one case, \nit developed as a subserosal mass in the uterus [8-10]. However, \nin none of the cases, polypoid endometriosis was associated with \ntamoxifen use, and this is the first report of tamoxifen treatment-\nassociated with polypoid endometriosis in Korea.\nConflict of interest\nNo potential conflict of interest relevant to this article was \nreported.\nAcknowledgments\nThis work was supported by the Soonchunhyang University \nresearch fund.\nReferences\n1. McCluggage WG, Bryson C, Lamki H, Boyle DD. Benign, \nborderline, and malignant endometrioid neoplasia arising \nin endometriosis in association with tamoxifen therapy. Int \nJ Gynecol Pathol 2000;19:276-9.\n2. Schlesinger C, Silverberg SG. Tamoxifen-associated pol -\nFig. 2. (A) An approximately 4×3-cm-sized, polypoid mass is noted in the right paratubal area. (B) At lower magnification, it appears as a multilocular cystic \nmass, which consists of cystically dilated endometrial glands and stroma (H&E, ×12.5). (C) The glands are lined by a single layer of columnar or cuboidal \ncells with no cellular atypia or mitosis (H&E, ×400). (D) The stromal cells show positivity for CD10 (CD10, ×200).\nA B\nCD\n\nwww.ogscience.org330\nVol. 58, No. 4, 2015\nyps (basalomas) arising in multiple endometriotic foci: a \ncase report and review of the literature. Gynecol Oncol \n1999;73:305-11. \n3. Chalas E, Costantino JP , Wickerham DL, Wolmark N, Lewis \nGC, Bergman C, et al. Benign gynecologic conditions \namong participants in the Breast Cancer Prevention Trial. \nAm J Obstet Gynecol 2005;192:1230-7.\n4. Kraft JK, Hughes T. Polypoid endometriosis and other \nbenign gynaecological complications associated with \nTamoxifen therapy-a case to illustrate features on mag -\nnetic resonance imaging. Clin Radiol 2006;61:198-201.\n5. Oxholm D, Knudsen UB, Kryger-Baggesen N, Ravn P . Post-\nmenopausal endometriosis. Acta Obstet Gynecol Scand \n2007;86:1158-64.\n6. Mostoufizadeh M, Scully RE. Malignant tumors arising in \nendometriosis. Clin Obstet Gynecol 1980;23:951-63.\n7. Parker RL, Dadmanesh F, Young RH, Clement PB. Polypoid \nendometriosis: a clinicopathologic analysis of 24 cases and a \nreview of the literature. Am J Surg Pathol 2004;28:285-97.\n8. Kim ES, Jeen YT, Lee D, Kim YJ, Park JY, Kim JN, et al. \nRectal polypoid endometriosis mimicking malignant rectal \ntumor. Korean J Gastrointest Endosc 2007;34:156-60.\n9. Park JI, Cho JH, Kim GR, Kim MJ. US or MR imaging fea -\ntures of polypoid endometriosis: a case report. J Korean \nSoc Radiol 2009;61:409-12.\n10. Cho YJ, Jeen YT, Kim YS, Chun HJ, Um SH, Kim CD, et \nal. Rectal endometriosis mimicking a large polypoid rectal \ncancer. Endoscopy 2006;38 Suppl 2:E48-9.","source_license":"CC0","license_restricted":false}