{"paper_id":"7f6ba2ed-480a-4974-b2da-83e2e4372323","body_text":"International Journal of Case Reports and Images, Vol. 13, 2022. ISSN: 0976-3198\nInt J Case Rep Images 2022;13:101302Z01NK2022.   \nwww.ijcasereportsandimages.com\nKumar et al. 1\nCASE REPORT PEER REVIEWED | OPEN ACCESS\nPolypoid endometriosis: A rare variant and a mimic of \novarian malignancy\nNeha Kumar, Nidhi Nayyar, Deepasha Garg, Rajan Duggal,  \nNarottam Khadaria\nABSTRACT\nIntroduction: Polypoid endometriosis is a rare \nform of endometriosis, distinct from classical or usual \nendometriosis. Unlike usual endometriosis which usually \npresents in young women, polypoid endometriosis occurs \nmore commonly in postmenopausal women. It mimics \nmalignancy and simulates ovarian cancer, not only on \npreoperative investigations but also intraoperatively, \ncausing a diagnostic dilemma.\nCase Report:  We describe here, a case of polypoid \nendometriosis that was presumed to be a case of ovarian \ncancer with peritoneal dissemination preoperatively. \nContrast enhanced computed tomography (CT) scan \nrevealed bilateral solid-cystic adnexal lesions, moderate \nascites, and peritoneal thickening. Serum CA-125 was \n1057 U/mL and serum HE4 was 88 pmol/L. Due to high \nindex of suspicion of malignancy, the patient was taken \nNeha Kumar 1, MBBS, MS, MCh, Nidhi Nayyar 2, MBBS, \nDNB, Deepasha Garg 3, MBBS, MD, Rajan Duggal 4, \nMBBS, MD, DNB, Narottam Khadaria 5, MBBS, MD\nAffiliations: 1Gynecologic Oncology, Senior Consultant, Gy -\nnecologic Oncology, Department of Surgical Oncology, BLK \nCancer Centre, BLK-Max Super Speciality Hospital, Delhi, \nIndia; 2Obstetrics & Gynecology, MNAMS, Fellowship in \nGynecologic Oncology, Associate Consultant, Gynecologic \nOncology, Department of Surgical Oncology, BLK Cancer \nCentre, BLK-Max Super Speciality Hospital, Delhi, India; \n3Pathology, Consultant, Histopathology & Cytopathology, \nBLK-Max Super Speciality Hospital, Delhi, India; 4Pathology, \nAssociate Director & HOD, Histopathology & Cytopathology, \nBLK-Max Super Speciality Hospital, Delhi, India; 5Senior \nConsultant, Radiology, Department of Radiology, BLK-Max \nSuper Speciality Hospital, Delhi, India.\nCorresponding Author: Dr. Neha Kumar, Senior Consultant, \nGynecologic Oncology, Department of Surgical Oncology, \nBLK Cancer Centre, BLK-Max Super Speciality Hospital, \nDelhi, India; Email: mail.nehakumar11@gmail.com\nReceived: 06 January 2022\nAccepted: 26 March 2022\nPublished: 27 April 2022\nfor exploratory laparotomy and underwent hysterectomy \nwith bilateral adnexectomy with excision of deposits \nover pouch of Douglas and both ureters and excision \nof adherent thickened omentum. Intraoperative frozen \nsection as well as the final histopathology was reported as \npolypoid endometriosis.\nConclusion: It is imperative for the gynecologist, \noncologist, and pathologist to know of this rare benign \nentity and distinguish it from ovarian malignancy, in \norder to avoid radical surgery and overtreatment. Surgery \nwith complete removal of all lesions is the cornerstone of \nmanagement of polypoid endometriosis and the patients \ngenerally have a good prognosis.\nKeywords: Cancer mimic, Endometriosis, Ovarian can-\ncer, Polypoid endometriosis\nHow to cite this article\nKumar N, Nayyar N, Garg D, Duggal R, Khadaria \nN. Polypoid endometriosis: A rare variant and a \nmimic of ovarian malignancy. Int J Case Rep Images \n2022;13:101302Z01NK2022.\nArticle ID: 101302Z01NK2022\n*********\ndoi: 10.5348/101302Z01NK2022CR\nINTRODUCTION\nPolypoid endometriosis is an uncommon variant of \nendometriosis, more commonly found in postmenopausal \nwomen, which mimics an ovarian malignancy both \npreoperatively and intraoperatively. It has histological \nfeatures akin to an endometrial polyp and most \ncommonly affects the ovaries, colon, mesentery, uterine \nserosa, cervical canal, vaginal mucosa, fallopian tubes, \nureters, bladder peritoneum, and paraurethral and \n\nInternational Journal of Case Reports and Images, Vol. 13, 2022. ISSN: 0976-3198\nInt J Case Rep Images 2022;13:101302Z01NK2022.   \nwww.ijcasereportsandimages.com\nKumar et al. 2\nparavaginal regions [1]. Here, we report a case of ovarian \npolypoid endometriosis that resembled ovarian cancer on \npreoperative clinical features and radiological imaging as \nwell as on the intraoperative gross findings. Intraoperative \nfrozen section and the final histopathology confirmed \nthis rare benign entity.\nCASE REPORT\nA 36-year-old woman with previous two normal \ndeliveries presented with complaints of abdominal \ndistension and bloating sensation for seven days. She \nhad past history of an endometriotic cyst 10 years back, \nfor which she received GnRH analogs for 3 months \nfollowed by laparoscopic left ovarian cystectomy. She was \nasymptomatic thereafter and was not on any follow-up or \nhormonal therapy for the same.\nThe ultrasound done at the time of presentation of \ncurrent complaints revealed a 10×6×5 cm solid cystic \nlesion in left adnexa with few calcific foci within it and \nmoderate ascites. The serum CA-125 and HE 4 values \nwere elevated 1057 U/mL and 88 pmol/L, respectively. \nRest of the tumor markers namely, carcinoembryonic \nantigen (CEA), CA-19.9, alpha-fetoprotein (AFP), Beta \nhuman chorionic gonadotropin (HCG), and lactate \ndehydrogenase (LDH) were within normal limits. Contrast \nenhanced CT scan of whole abdomen showed solid \ncystic mass lesions in bilateral adnexa (left 11.3×7.1×8.8 \ncm; right 5.0×3.4×5.8 cm) extending into pouch of \nDouglas, bilateral tiny anterior sub-diaphragmatic lymph \nnodes, moderate free fluid in abdomen, and peritoneal \nthickening (Figure 1A and B).\nIn view of high index of suspicion for malignancy, \nthe patient was taken up for exploratory laparotomy. \nIntraoperative findings showed a 10×7 cm polypoidal, \nexophytic left adnexal mass adherent to left ureter, bulky \nright ovary with few cysts on ovarian surface, deposits \npresent over uterus, both ureters, ovarian fossae, \nappendix and pouch of Douglas, thickened omentum \nand around 750 mL of serosanguinous ascitic fluid. Left \nadnexal mass was excised and sent for frozen section \nwhich was reported as polypoidal endometriosis (Figure \n2A).\nType I hysterectomy with bilateral adnexectomy with \nadhesiolysis, excision of deposits over pouch of Douglas \nand both distal ureters, excision of adherent thickened \nomentum, appendectomy, and bilateral DJ stenting \nwas done. Final histopathological report was suggestive \nof polypoidal endometriosis presenting as left adnexal \nmass and causing extensive hemorrhagic deposits in \npouch of Douglas, left parametrium and right and left \nureters with endometriosis of right adnexa (Figure 2B). \nImmunohistochemistry was positive for estrogen receptor \n(ER) and CD10, ER highlighting the benign endometrial \nglands and CD10 the surrounding endometrial stroma. \nThe postoperative period was uneventful and bilateral \nDJ-stents were removed after six weeks. The patient \nreceived two doses of injection Leuprolide Depot 11.25 \nmg, and has had follow-up visits till 18 months post-\nsurgery. She has been disease-free and symptom-free till \npresent.\nFigure 1: Contrast-enhanced CT scan (A: axial section, B: \ncoronal section) showing left adnexal mass, bulky right adnexa, \nascites, and peritoneal thickening.\nFigure 2: (A) Photo panel shows hemorrhagic polypoidal \ntissue fragment in gross morphology. (B) Microscopy shows \npolyp composed of few cystically dilated endometrial glands \nwith intervening fibrocellular stroma. There is no evidence \nof dysplasia or invasive malignancy. Hematoxylin & Eosin, \noriginal magnification × 20.\nDISCUSSION\nPolypoid endometriosis is a rare variant of \nendometriosis presenting as polypoidal mass(es) which \nmay be misinterpreted as a neoplasm, preoperatively on \nclinical examination and imaging, intraoperatively on \ngross examination, and postoperatively on pathological \nassessment. It occurs over a wide age range (23–78 years) \nbut more commonly in the postmenopausal women (mean \nage 52.5 years), unlike usual or classical endometriosis \nwhich is commoner in younger women [1]. The usual \nsymptoms are abdominal pain, vaginal bleeding, and \nurinary symptoms like frequency, hematuria, and \nurgency. The clinical presentations include pelvic \nmass, vaginal polypoid mass, large bowel obstruction, \nor an incidental finding in hysterectomy with bilateral \nsalpingo-oophorectomy specimen. The most common \nsites of involvement are large bowel and its mesentery, \novary, uterine serosa, vaginal and cervical mucosa, \nureter, fallopian tube, omentum, bladder, paraurtehral \nand paravaginal regions, and the retroperitoneum [1].\nThe term “polypoid endometriosis” was coined \nby Mostofizadeh and Scully for a distinct type of \n\nInternational Journal of Case Reports and Images, Vol. 13, 2022. ISSN: 0976-3198\nInt J Case Rep Images 2022;13:101302Z01NK2022.   \nwww.ijcasereportsandimages.com\nKumar et al. 3\nendometriosis with histopathological features simulating \nan endometrial polyp [2]. Nearly half the cases occur \nin a milieu of exogenous hormone intake, including \nhormone replacement therapy (unopposed estrogen \nor mixed estrogen-progestin), tamoxifen (which acts \nas an estrogen agonist on endometrium) or after the \nwithdrawal of GnRH analogs (used in the treatment of \nusual endometriosis) [1, 3–5].\nThe imaging of choice is an magnetic resonance \nimaging (MRI) where the lesion appears as a T2 \nhyperintense polypoidal mass with a T2 hypointense \nperipheral rim. On contrast enhancement, the pattern \nappears similar to that of endometrium and there is lack \nof diffusion restriction [6].\nIn this case, the patient was a young 36-year-old \nwoman, with prior history of use of GnRh analogs for \nthree months followed by left ovarian cystectomy for \nendometriotic cyst done 10 years back. Although MRI is \nthe imaging of choice, there was a high index of suspicion \nfor malignancy in this case, with the ultrasound showing \na large solid cystic adnexal mass and moderate ascites, \nand hence a contrast CT scan of the whole abdomen \nwas ordered. The benign nature of the left adnexal mass \non frozen section report was informed to the patient’s \nrelatives, but they opted for removal of the bulky right \novary and uterus instead of only right ovarian cystectomy \nfollowed by close surveillance.\nHistopathological presentation of polypoid \nendometriosis includes presence of polypoid tumor-like \nmasses projecting from a serosal or mucosal surface \nor the lining epithelium of an endometriotic cyst. On \ngross examination, the lesions are fleshy with cystic \nchanges and hemorrhage. Microscopic examination \nshows endometrial glands and stroma with a variety of \narchitectural patterns in glandular epithelium. The most \ncommon glandular pattern is simple cystic and noncystic \nhyperplasia without atypia. Other patterns include \nsimple or complex hyperplasia with atypia, disordered \nproliferation, and cystic atrophy. Epithelial metaplasia \n(tubal, mucinous, papillary, and squamous), hemorrhage, \nfibrosis, hemosiderin laden histiocytes, and decidual \nchanges are also present. In some cases, the lesion may \nresemble an intrauterine endometrial polyp. Stromal \ncell atypia is generally absent and most of the cases \nare found in the proliferative phase stroma. The main \ndifferential diagnosis in a case of polypoid endometriosis \nis a mullerian adenosarcoma. Unlike adenosarcoma, \npolypoid endometriosis lacks periglandular stromal \nhypercellularity, stromal papillae, and stromal atypia.\nIn the largest series of 24 cases of polypoid \nendometriosis reported by Parker et al., synchronous or \ncontiguous usual type endometriosis was found in 75% \nof cases and/or previous history of endometriosis in 29% \ncases [1]. Polypoid endometriosis is generally associated \nwith a benign course and surgery with complete removal \nof all the lesions is the key to the management of the \ndisease.\nCONCLUSION\nPolypoid endometriosis is a distinct and rare variant \nof endometriosis which may simulate a neoplasm on \nclinical presentation, imaging, intraoperative findings, \nand histopathological evaluation. Unlike classical or \nusual endometriosis, it is commoner in postmenopausal \nwomen, although it may occur over a wide age range. \nIt usually occurs in the milieu of exogenous hormone \nintake. Surgery with complete removal of all lesions is \nthe cornerstone of management of the disease. In spite \nof presence of hyperplastic or metaplastic glands and \noccasional cytological atypia, the disease follows a benign \ncourse and progression to neoplasm, if any, is rare. \nAlthough it is an uncommon entity, the gynecologists and \npathologists should be aware of this form of endometriosis \nsince the clinical, radiological as well as the intraoperative \nfindings resemble that of malignancy, and being a benign \nmimic, it can be treated with a conservative surgery \nwith excision of lesions rather than the radical surgery \n(staging/cytoreduction) done for ovarian cancer.\nREFERENCES\n1. Parker RL, Dadmanesh F, Young RH, Clement PB. \nPolypoid endometriosis: A clinicopathologic analysis \nof 24 cases and a review of the literature. Am J Surg \nPathol 2004;28(3):285–97.\n2. Mostoufizadeh M, Scully RE. Malignant tumors \narising in endometriosis. Clin Obstet Gynecol \n1980;23(3):951–63.\n3. Jaegle WT, Barnett JC, Stralka BR, Chappell NP. \nPolypoid endometriosis mimicking invasive cancer in \nan obese, postmenopausal tamoxifen user. Gynecol \nOncol Rep 2017;22:105–7.\n4. Choi IH, Jin SY, Jeen YM, Lee JJ, Kim DW. \nTamoxifen-associated polypoid endometriosis \nmimicking an ovarian neoplasm. Obstet Gynecol Sci \n2015;58(4):327–30.\n5. Othman NH, Othman MS, Ismail AN, Mohammad \nNZ, Ismail Z. Multiple polypoid endometriosis \n– A rare complication following withdrawal of \ngonadotrophin releasing hormone (GnRH) agonist \nfor severe endometriosis: A case report. Aust N Z J \nObstet Gynaecol 1996;36(2):216–8.\n6. Ghafoor S, Lakhman Y, Park KJ, Petkovska I. Polypoid \nendometriosis: A mimic of malignancy. Abdom Radiol \n(NY) 2020;45(6):1776–82. \n*********\nAuthor Contributions\nNeha Kumar – Conception of the work, Design of the \nwork, Acquisition of data, Analysis of data, Interpretation \nof data, Drafting the work, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \n\nInternational Journal of Case Reports and Images, Vol. 13, 2022. ISSN: 0976-3198\nInt J Case Rep Images 2022;13:101302Z01NK2022.   \nwww.ijcasereportsandimages.com\nKumar et al. 4\nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nNidhi Nayyar – Design of the work, Acquisition of \ndata, Analysis of data, Interpretation of data, Drafting \nthe work, Revising the work critically for important \nintellectual content, Final approval of the version to be \npublished, Agree to be accountable for all aspects of the \nwork in ensuring that questions related to the accuracy \nor integrity of any part of the work are appropriately \ninvestigated and resolved\nDeepasha Garg – Acquisition of data, Analysis of data, \nInterpretation of data, Revising the work critically for \nimportant intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nRajan Duggal – Acquisition of data, Analysis of data, \nInterpretation of data, Revising the work critically for \nimportant intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nNarottam Khadaria – Acquisition of data, Analysis of \ndata, Interpretation of data, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nGuarantor of Submission\nThe corresponding author is the guarantor of submission.\nSource of Support\nNone.\nConsent Statement\nWritten informed consent was obtained from the patient \nfor publication of this article.\nConflict of Interest\nAuthors declare no conflict of interest.\nData Availability\nAll relevant data are within the paper and its Supporting \nInformation files.\nCopyright\n© 2022 Neha Kumar et al. 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