{"paper_id":"6a991184-1f52-47c3-acb1-b56f61ec9832","body_text":"DIANA et al. Obstet Gynecol Cases Rev 2023, 10:243\nVolume 10 | Issue 3\nDOI: 10.23937/2377-9004/1410243\nCitation: DIANA C, Bursać D, Perković P, Duić Ž,Gašparov S (2023) Atypical Sites of Endometriosis: Pri-\nmary Inguinal Cutaneous Endometriosis. Obstet Gynecol Cases Rev 10:243. doi.org/10.23937/2377-\n9004/1410243\nAccepted: July 03, 2023: Published: July 05, 2023\nCopyright: © 2023 DIANA C, et al. This is an open-access article distributed under the terms of the \nCreative Commons Attribution License, which permits unrestricted use, distribution, and reproduction \nin any medium, provided the original author and source are credited.\n• Page 1 of 3 •\nDIANA et al. Obstet Gynecol Cases Rev 2023, 10:243\nOpen Access\nISSN: 2377-9004\nObstetrics and\nGynaecology Cases - Reviews\nAtypical Sites of Endometriosis: Primary Inguinal Cutaneous \nEndometriosis\nCULEJ DIANA1*, Bursać D1,2, Perković P1, Duić Ž1,3 and Gašparov S3,4\n1Department of Obstetrics and Gynecology, University Hospital Merkur, Zagreb, Croatia\n2University of North, University Center Varaždin, Croatia\n3School of Medicine, University of Zagreb, Croatia\n4Department of Pathology, University Hospital Merkur, Zagreb, Croatia\nCASE  REpoRt\nAbstract\nEndometriosis is a chronic inflammatory disease defined \nas the presence of endometrium-like tissue outside the \nuterus. It is mostly found in women of reproductive age. \nDepending on the involved site, women can present with \nvarious symptoms. We report a case of rare, atypical site of \nendometriosis - primary inguinal cutaneous endometriosis. \nA 27-year-old woman presented to our Deparment with a \nhistory of a painful, firm, movable, subcutaneous nodule \n1 × 1 cm of size in the right inguinum. She experienced \nflares of pain with her menstrual cycle and dysmenorrhea. \nUltrasound revealed hypoechoic nodule with vascular flow. \nPunch biopsy of the nodule was performed. The biopsy \nresults showed adipocytes and cell inclusions partially \npositive on epithelial membrane antigen. After surgical \nexcision of nodule with wide margins, tissue was consisted \nfrom endometrial glands with encompassing fibrotic stroma. \nNo additional signs of endometriosis were found. Because \nthe condition is rare and can mimic presentations of other \ndiseases, such as lymphatic nodule, keloid, dermatofibroma \nprotuberans, cutaneous metastasis of cancer or \ndermatofibroma, cutaneous endometriosis can be difficult to \ndiagnose.\nKeywords\nInguinal cutaneous endometriosis, Primary endometriosis, \nEpithelial membrane antigen\nCheck for\nupdates\noutside the uterus [1]. It is mostly found in women of \nreproductive age, within the general female population \nfrom 2 to 10%, up to 50% in infertile women [1]. \nEndometriosis lesions can be classified as ovarian, \nexclusively extra-ovarian or mixed [1-3]. The common \nsites of endometriosis are the ovaries, fallopian tubes, \npelvic peritoneum and uterosacral ligaments, whereas \nthe atypical sites include the gastrointestinal tract, \nurinary tract, soft tissues, chest etc [1-4]. Depending \non the involved site, women can present with various \nsymptoms. Most common are deep pelvic pain, \ndyspareunia and dysmenorrhea, but can be bowel \nobstruction, melena, hematuria, dysuria, dyspnea and \nswelling in soft tissues [1-4]. The average between \nonset of symptoms and diagnosis is between 8-12 years \n[1]. Diagnosis of extra-pelvic endometriosis can be \ndemanding.\nCase Description\nWe report a case of rare, atypical site of endometriosis \n- primary inguinal cutaneous endometriosis. It is \nrelatively uncommon and occurs when endometrial \nglands and stroma reside in the skin. Cutaneous \nendometriosis can be divided into primary and secondary \nendometriosis, depending on past medical history. A \n27-year-old woman, gravida 0, para 0, presented to our \nGynecology Deparment with flares of sharp, focal pain \nin inguinal region and connected it with her menstrual \n*Corresponding author: Culej Diana, MD, Department of Obstetrics and Gynecology, University Hospital Merkur, Zajčeva \n19, 10 000 Zagreb, Croatia, Tel: +385981931909\nIntroduction\nEndometriosis is a chronic inflammatory disease \ndefined as the presence of endometrium-like tissue \n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410243\nDIANA et al. Obstet Gynecol Cases Rev 2023, 10:243\n• Page 2 of 3 •\nis a rare disorder that can mimic presentations of \nother diseases. If is suspected, a punch biopsy can be \ncycle. Her past gynecological medical history revealed \ndysmenorrhea. Results of laboratory evaluation and \nof tumor markers were within the reference values. \nShe was able to palpate a tender mass arising from \nher right inguinum. On physical examination, a firm, \npainfull, movable subcutaneous nodule 1 × 1 cm of \nsize with slight erythema of the overlying skin was \nobserved. Ultrasound revealed hypoechoic nodule \nwith vascular flow. Punch biopsy of the nodule was \nperformed. The biopsy results showed adipocytes and \ncell inclusions partially positive on epithelial membrane \nantigen. After surgical excision of the nodule with wide \nmargins, histopathological analysis demonstrated that \nthe tissue was consisted from endometrial glands with \nencompassing fibrotic stroma. A routine gyecologic \nexamination was unremarkable. Abdominal and \ntransvaginal ultrasond showed no abnormalities, as well \nas pelvic magnetic resonance imaging. No additional \nsigns of endometriosis were found.\nDiscussion\nCutaneous endometriosis represents 0.5-1.0% of \npatients with atypical site of endometriosis [1, 4]. It is \noften associated with prior surgical medical history \n[1,4]. If it presents without it, it is known as primary \nspontaneous cutaneous endometriosis [1-6]. From \nall extrapelvic endometriosis, umbilical occupies over \n40%, followed by the inguinal area, perineal region and \nabdominal wall [4, 6]. The mean age at presentation is \n34 years [7]. The clinical diagnosis relies mostly on the \nrecognition of the cyclic symptoms of pain and swelling \nof the lesion [1-6]. Almost 25% of women with cutaneous \nendometriosis also experience intrapelvic endometriosis \n[6], although our patient did not have any signs of it. \nOriginates due to reflux of menstrual tissue through the \noviducts, mechanical placement of endometrial tissue \npost-abdominal operation, presence of embryonic \nrests, peritoneal mesothelial cells becoming metaplastic \n(celomic metaplastic theory) or lymphatic and vascular \nspread [1,7]. The most commonly accepted pathogenesis \nare lymphatic or vascular migration due to migration of \nendometrial tissue from retrogression of menstruation \n[1,4,6]. A punch biopsy is not commonly used to \ndiagnose endometriosis, however, if performed the \nresults may show the presence of epithelial membrane \nantigen, CD 10, estrogen, progesteron receptors or Ki67 \n[7,8]. Cutaneous endometriosis is treated with surgical \nresection [1, 4,6]. Histopathological examination is \ncrucial and major discovery is that the tissue is consisted \nfrom endometrial glands with encompassing fibrotic, \nspindle-cell apperance stroma [7,8]. Primary cutaneous \nendometriosis can be differentially diagnosed with \nlymphatic nodule, keloid, dermatofibroma protuberans, \ncutaneous metastasis of cancer or dermatofibroma \n[2,6] (Figure 1, Figure 2 and Figure 3).\nConclusion\nWe conclude that primary cutaneous endometriosis \n         \nFigure 1: Microscopic picture of the lesion showing \nendometrial glands and stroma in the subcutaneous tissue \nof the skin (H&E x20). \n         \nFigure 2: Microscopic picture of the lesion showing \nendometrial glands and stroma in the subcutaneous tissue \nof the skin (H&E x40). \n         \nFigure 3: Microscopic picture of the lesion showing \nendometrial glands and stroma in the subcutaneous tissue \nof the skin (CD10).\n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410243\nDIANA et al. Obstet Gynecol Cases Rev 2023, 10:243\n• Page 3 of 3 •\n2. Raffi L, Suresh R, McCalmont TH, Twigg AR (2019) \nCutaneous endometriosis. Int J Womens Dermatol 5: 384-\n386.\n3. Lopez-Soto A, Sanchez-Zapata MI, Martinez-Cendan \nJP, Reina SO, Manas CM, et al. (2018) Cutaneous \nendometriosis: Presentation of 33 cases and literature \nreview. Eur J Obstet Gynecol Reprod Biol 221: 58-63.\n4. Loh SH, Lew BL, Sim WY (2017) Primary cutaneous \nendometriosis of umbilicus. Ann Dermatol 29: 621-625.\n5. Zhai J (2014) Spontaneous cutaneous endometriosis in the \nmons pubis region: A case report diagnosed by fine-needle \naspiration biopsy. Diagn Cytopathol 42: 615-618.\n6. Davis AC, Goldberg JM (2017) Extrapelvic endometriosis. \nIn: Seminars in reproductive medicine 2017 Jan. Thieme \nMedical Publishers.\n7. Farooq U, Laureano AC, Miteva M, Elgart GW \n(2011) Cutaneous endometriosis: Diagnostic \nimmunohistochemistry and clinicopathologic correlation. \nJournal of Cutaneous Pathology 38: 525-528.\n8. Tidman MJ, MacDonald DM (1988) Cutaneous \nendometriosis: A histopathologic study. Journal of the \nAmerican Academy of Dermatology 18: 373-377.\nperformed, but surgical excision of the affected tissue \nand histopathological analysis is the golden standard. \nThis implies that the diagnosis is difficult and requires \na great deal of effort, skill and expertise to identify and \nconfirm.\nAcknowledgements\nNone.\nSources of Support\nNone.\nStatement of Equal Authors Contribution\nThe authors declare that they equally contributed \nregarding the publication of this paper.\nReferences\n1. Becker CM, Bokor A, Heikinheimo O, Horne A, Jansen \nF, et al. (2022) ESHRE guideline: Endometriosis. Human \nreproduction open.","source_license":"CC0","license_restricted":false}