{"paper_id":"cf7195a1-f54d-4d50-b725-6bac91fb8b8b","body_text":"Journal of Case Reports and Images in Obstetrics and Gynecology, Volume 9, Issue 1, 2023; Pages 11–16. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2023;9(1):11–16. \nwww.ijcriog.com\nBeroukhim et al. 11\nCASE REPORT  OPEN ACCESS  \nSurgical management of inguinal endometriosis:  \nCase report and surgical video\nGabriela Beroukhim, Ecem Esencan, Padmini Manrai,  \nMasoud Azodi, Yonghee K Cho\nABSTRACT\nIntroduction: Inguinal endometriosis is a rare type \nof extra-pelvic endometriosis, which may occur in the \nabsence of symptoms of intra-pelvic endometriosis. This \ncase report highlights the importance of considering \ninguinal endometriosis in the workup of an inguinal mass \nand demonstrates a step-by-step surgical approach to \nmanagement, with an accompanying video.\nCase Report:  We encountered a case of a 31-year-\nold nulligravid woman who presented with a painful \nright inguinal mass. The patient underwent diagnostic \nlaparoscopy, which was notable for Stage 1 intra-pelvic \nendometriosis, without involvement of the internal \ninguinal ring or round ligament. The inguinal mass was \ncarefully resected from nearby vessels, muscles, and \nnerves. Pathology confirmed endometriosis.\nConclusion: Gynecologists, in collaboration with a \nmultidisciplinary team, should be prepared to workup, \ndiagnose, and surgically manage inguinal endometriosis. \nGabriela Beroukhim 1, Ecem Esencan 1, Padmini Manrai 2,  \nMasoud Azodi 3, Yonghee K Cho 4\nAffiliations: 1MD, Resident Physician, Department of Ob -\nstetrics, Gynecology & Reproductive Sciences, Yale School \nof Medicine, New Haven, CT, USA; 2MD, Gynecologic Pa -\nthology Fellow, Departments of Pathology and Laboratory \nMedicine, Yale School of Medicine, New Haven, CT, USA; \n3MD, Professor; Director of Minimally Invasive and Robotic \nSurgery, Director of Minimally Invasive Gynecologic Surgery \nFellowship, Department of Obstetrics, Gynecology & Repro-\nductive Sciences, Yale School of Medicine, New Haven, CT, \nUSA; 4MD, FACOG, Assistant Professor, Department of Ob-\nstetrics, Gynecology & Reproductive Sciences, Yale School \nof Medicine, New Haven, CT, USA.\nCorresponding Author: Gabriela Beroukhim, MD, 20 York \nStreet, New Haven, CT 06511, USA; Email: gabrielaberouk-\nhim@gmail.com\nReceived: 12 April 2022\nAccepted: 25 June 2022\nPublished: 07 February 2023\nWhen this condition is suspected, imaging should be \nobtained, and tissue biopsy may be considered, provided \nthat a hernia has been ruled out. Surgical management \nis typically recommended and should entail diagnostic \nlaparoscopy and excisional surgery.\nKeywords: Extra-pelvic endometriosis, Inguinal endo -\nmetrioma, Inguinal mass\nHow to cite this article\nBeroukhim G, Esencan E, Manrai P, Azodi M, Cho \nYK. Surgical management of inguinal endometriosis:  \nCase report and surgical video. J Case Rep Images \nObstet Gynecol 2023;9(1):11–16.\nArticle ID: 100136Z08GB2023\n*********\ndoi: 10.5348/100136Z08GB2023CR\nINTRODUCTION\nEndometriosis is a chronic disease, affecting 5–10% \nof reproductive-age women globally, characterized by \nendometrial-like tissue present outside of the uterus \n[1]. The most common sites of endometriosis are within \nthe pelvis (ovaries, anterior and posterior cul-de-sac, \novarian fossa, posterior broad ligaments, uterosacral \nligaments, uterus, fallopian tubes, and round ligament) \n[2]. Less commonly, endometriosis involves the small \nand large bowel, appendix, bladder, ureters, vagina, \ncervix, rectovaginal septum, inguinal canals, umbilicus, \nand surgical scars [2]. Rare reports of extra-pelvic \nendometriosis in the breast, pancreas, liver, gallbladder, \nkidney, urethra, extremities, vertebrae, bone, peripheral \nnerves, spleen, diaphragm, central nervous system, \nhymen, and lung have been reported [1].\nCASE REPORT  PEER REVIEWED | OPEN ACCESS \n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 9, Issue 1, 2023; Pages 11–16. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2023;9(1):11–16. \nwww.ijcriog.com\nBeroukhim et al. 12\nInguinal endometriosis affects 0.3–0.6% of women \nwith endometriosis [3, 4]. The etiology is not well \nunderstood, but it may develop in the inguinal region as \na mass from direct implantation, coelomic metaplasia, \ntubal regurgitation, and/or lymphatic spread, though \nall theories are disputable [3]. It has been reported \nto develop predominantly on the right side  and often \npresents as swelling and/or pain in the inguinal region \nduring menstruation [4, 5]. Inguinal endometriosis can \nbe difficult to diagnose and may be confused for hernia, \nlymphadenopathy, abscess, neuroma, hydrocoele of the \ninguinal canal, primary or metastatic cancer, lipoma, \nhematoma, sarcoma, and subcutaneous cyst [6]. In the \ncase presented herein, we describe the presentation, \nworkup, and surgical management of a woman with \ninguinal endometriosis. This case report is accompanied \nby a surgical video demonstrating the stepwise approach \nfor surgical resection of inguinal endometriosis.\nCASE REPORT\nA 31-year-old nulligravid woman with medical history \nnotable for cerebral palsy and without history of intra-\nabdominal surgery presented for an annual well-woman \nvisit with a painful, right inguinal mass. On initial \nevaluation, the <1 cm mildly tender lesion was suspected \nto be an ingrown hair. Over the course of one year, the \npatient represented, reporting that the mass had become \n“deeper, firmer, and larger.” Physical examination at the \ntime was notable for a 3-cm non-mobile firm mass in her \nright inguinal fold. The patient reported that the pain \nwas intermittent, occurring multiple times throughout \nthe day, and not related to her menses. She had no \nsymptoms of intra-pelvic endometriosis, including history \nof dysmenorrhea, chronic pelvic pain, abdominal pain, \ndyspareunia, dyschezia, or dysuria. The patient had never \nused hormonal suppression throughout her reproductive \nlifetime. A computed tomography (CT) of the abdomen \nand pelvis was initially obtained for consideration of an \ninguinal hernia and was notable for an irregular 2–3 cm \nsoft tissue nodule in the right inguinal region (Figure 1). \nSubsequent magnetic resonance imaging (MRI) of the \nabdomen and pelvis was obtained to further characterize \nthe nodule and revealed a right inguinal 2–3-cm ill-\ndefined, soft tissue lesion adherent to the fascia. The lesion \nwas hypointense on T2-weighted sequence and moderately \nintense lesion with a few foci of hyperintense signal on T1-\nweighted sequence, favoring endometriosis (Figure 2). \nThe visceral pelvis was without evidence of endometriosis \non imaging.\nGeneral surgery and gynecologic oncology were \nconsulted preoperatively given the broad differential \ndiagnosis, which included malignancy, and due to the \nlocation of the lesion. A core biopsy of the mass by a \ngeneral surgeon was notable for microscopic foci of benign \nendometrial glandular tissue with stroma as well as fibrosis \nand hemosiderin deposits, confirming endometriosis. \nThe patient was offered expectant, medical in the form of \nhormonal suppression, or surgical management, to include \ndiagnostic laparoscopy, to assess for intra-abdominal \nendometriosis, as well as excisional surgery. The patient \nopted for surgical management.\nDiagnostic laparoscopy was performed to assess for \nthe presence and degree of abdominal endometriosis, \nwith specific consideration paid to evaluating the round \nligament and internal inguinal ring to ensure a clear margin \nand complete excision of the inguinal endometrioma. \nLaparoscopy revealed Stage I intra-pelvic endometriosis, \nwith less than 1-cm lesions on the left ovary and uterosacral \nligament. The round ligament and internal inguinal ring \nwere assessed for involvement with the inguinal mass and \nwere noted to be without evidence of endometriosis. In \nthe inguinal region, an incision was made at the inguinal \nligament and taken down to the superficial fascia using \nelectrocautery to minimize bleeding. The mass was \ndensely adhered to the pubic symphysis and external \noblique aponeurosis. Moreover, the mass appeared to \ninvolve adjacent vessels, nerves, and muscles. The mass \nFigure 1: CT of the abdomen and pelvis with evidence of a right \ninguinal soft tissue lesion. A sagittal view of the pelvis with an \nirregular 2–3 cm soft tissue nodule in the right inguinal region. \nThere is no evidence of inguinal hernia.\nFigure 2: MRI of the abdomen and pelvis with a right-sided \ninguinal lesion concerning for endometriosis. A sagittal view of \nthe pelvis with a right inguinal 2–3-cm ill-defined, soft tissue \nlesion adherent to the fascia. (A) On T2-weighted sequence the \nlesion is hypointense. (B) On T1-weighted sequence the lesion \nis intermediate with a few foci of hyperintense signal favoring \nendometriosis.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 9, Issue 1, 2023; Pages 11–16. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2023;9(1):11–16. \nwww.ijcriog.com\nBeroukhim et al. 13\nwas progressively mobilized from the superficial inguinal \nring superiorly, sartorius muscle laterally, and adductor \nlongus muscle medially. The superficial epigastric \nvessel and several perforating branches of the femoral \nvein (including the saphenous, superficial epigastric, \nand superficial external pudendal veins) as well as the \nround ligament of the uterus, at the level of the external \ninguinal ring, were ligated and tied to free the mass from \nthe surrounding tissue. Due to tumor involvement, the \nilioinguinal nerve was intentionally cut. The mass was \nremoved en bloc, and the resected bed fulgurated to ablate \nresidual microscopic lesions of endometriotic tissue. The \nintegrity of the abdominal wall was assessed by a general \nsurgeon. The facial defect was adequately repaired via a \nprimary closure in multiple layers, without the need for a \nmesh. The technical steps of management and resection of \nan inguinal endometrioma have been detailed in the video \nwith an emphasis on anatomic landmarks by utilizing \nvisual illustrations (Video 1). Final pathology confirmed \nextensive inguinal endometriosis, characterized by \nfoci of benign endometrial glands and stroma within \nsmooth muscle and fibroadipose tissue. One lymph node \naccompanying the specimen also demonstrated focal \nendometriosis (Figure 3). Positive CD10 immunostaining \nhighlighting the endometrial stroma supported the \ndiagnosis.\nThe patient was discharged home on the day of surgery. \nHer postoperative course was uncomplicated. She was \nevaluated one month postoperatively and reported no \nresidual inguinal pain. The patient had no perceived \nresidual nerve deficit or neuropathy.\nVideo 1: Surgical resection of inguinal endometriosis. The \ntechnical steps of management and resection of an inguinal \nendometrioma are detailed in the surgical video. Diagnostic \nlaparoscopy was initially performed and revealed Stage I intra-\npelvic endometriosis; notably, the round ligament and internal \ninguinal ring were without evidence of endometriosis. An incision \nwas then made at the inguinal ligament and taken down to the \nsuperficial fascia using electrosurgery (coagulation). The mass \nwas progressively mobilized from adherent adjacent structures. \nThe superficial epigastric vessel and several perforating branches \nof the femoral vein (including the saphenous, superficial \nepigastric, and superficial external pudendal veins) as well as the \nround ligament of the uterus, at the level of the external inguinal \nring, were ligated and tied. The mass was removed en bloc. The \nincision was then closed in multiple layers.\nVideo 1 URL: https://www.idoriums.com/edpanel/preview_\narticle/100136Z08GB2023#video1\nAccess Video \non other devices\nFigure 3: Inguinal endometriosis involving a lymph node. (A) \nLow power view demonstrating the two components necessary \nfor histologic diagnosis of endometriosis, endometrial glands \naccompanied by endometrial stroma (4×). (B) High power view \nbetter delineating the endometrial stroma that immediately \nsurrounds the glandular epithelium from the background lymph \nnode (10×). (C) fibrotic tissue with endometriosis (4×).\nDISCUSSION\nInguinal endometriosis is a rare diagnosis of extra-\npelvic endometriosis, characterized by endometriotic \nlesions in the round ligament, lymph nodes, subcutaneous \nadipose tissue, or hernias of the inguinal regions [2]. \nThree clinical types of inguinal endometriosis have been \ndescribed: type I lesions are located at a hernia sac or \nhydrocele of Nuck’s canal, type II lesions are on the round \n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 9, Issue 1, 2023; Pages 11–16. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2023;9(1):11–16. \nwww.ijcriog.com\nBeroukhim et al. 14\nligament, and type III lesions are located under the skin \n[5]. Inguinal endometriosis may be commonly mistaken \nfor, or occur concomitantly with, an inguinal hernia [5].\nThe diagnosis of inguinal endometriosis is difficult, \nwith the correct preoperative diagnosis made less than \n50% of the time [7]. Patients with inguinal endometriosis \ntypically present with swelling and/or pain in the \ninguinal region [4, 5]. In a systematic review of extra-\npelvic endometriosis, including 230 cases of parietal \nendometriosis (133 in the groin, 82 umbilical, 13 \nabdominal wall, and two perineal), symptoms included \na palpable mass (99%), cyclic pain (71%), and cyclic \nbleeding (48%) [1]. Differentiating whether the pain \nis temporally related to menses can be important in \ndistinguishing endometriosis from other pathologies. \nUp to 91% of inguinal endometriosis cases are associated \nwith coexisting pelvic endometriosis [3], though it may \noccur in the absence of pelvic endometriosis or pelvic \npain [6, 8]. The majority of cases are associated with a \nhistory of abdominal surgery or trauma [6].\nThe diagnostic workup of inguinal endometriosis \nshould entail a physical examination and imaging (such \nas ultrasound, CT, or MRI) [3]. Ultrasound is typically \nused as a first line imaging modality, as it may be used \nto rule out hernia. However, the appearance of inguinal \nendometriosis on ultrasound may be variable, including \nsolid masses, cystic masses, and combined cystic and \nsolid masses [9]. Magnetic resonance imaging can be \nparticularly useful for identifying distinct characteristics, \nsuch as the presence of iron in the hemosiderin deposits \ncontained in hemorrhagic micro-cysts in T1-weighted \nimages [10]. Biopsy or fine needle aspiration may be \nperformed, provided that a diagnosis of hernia is ruled \nout, and is useful for confirmation of the diagnosis.\nOnce the diagnosis is confirmed, patients may be \noffered expectant, medical, or surgical management. \nSurgical management by radical excision of the lesion and \nextraperitoneal round ligament is currently considered \nthe favored treatment option [11]. The literature supports \nat least 1-cm margins to ensure complete resection of the \nmass and decrease risk of recurrence [12]. In a systematic \nreview of extra-pelvic, parietal endometriosis, patients \nunderwent surgical management in 97% (222/227) of \ncases, 99% of which involved wide local excision, with \n5% recurrence and no complications [1]. In the same \nobservational study, general surgeons performed in 71.1% \n(158/222) and gynecologists in 15.7% (35/222) of cases. \nA multidisciplinary approach to surgical management \nshould be employed to optimize patient outcomes, \nwith involvement of general surgery, gynecology, \nand potentially gynecologic oncology, as was done in \nthis case [1]. Only two cases of laparoscopic excision \nof inguinal endometriosis have been reported [1]. In \ngeneral, diagnostic laparoscopy at the time of inguinal \nendometriosis excision is recommended [2, 3, 13], as \ninguinal endometriosis is frequently found in the setting \nof pelvic endometriosis, which as considerable sequelae. \nFurthermore, diagnostic laparoscopy may be necessary \nto rule out involvement of the internal inguinal ring and \nround ligament to achieve appropriate negative margins. \nHormonal therapy may be recommended subsequent to \nsurgery, as it may play a role in preventing recurrence [3].\nEvidence-based approaches regarding treatment \noptions and outcomes of inguinal endometriosis \nremain enigmatic given the low prevalence of inguinal \nendometriosis and the limited quantity and quality \nof data available. Observant management should be \npreferentially reserved for asymptomatic patients. \nHormonal treatment has been underreported as a \ntherapeutic strategy for inguinal endometriosis [14, \n15], and is typically reserved for patients who decline \nsurgical management. In a small retrospective study, oral \ncontraceptives were effective in symptomatic treatment \nof 1 of 4 patients without significant adverse effects, \nwhereas dienogest effectively improved pain in 6 of 7 \ncases [4]. Importantly, neither observant nor medical \nmanagement will result in resolution of the mass.\nCONCLUSION\nIn conclusion, inguinal endometriosis is a rare \nexample of extra-pelvic endometriosis. When inguinal \nendometriosis is suspected, we recommend imaging and \nconsideration of tissue biopsy (provided the diagnosis \nof a hernia has been ruled out), as the diagnosis can be \ndifficult to make. We recommend a multidisciplinary \napproach to treatment, particularly between gynecologists \nand general surgeons, given the proximity to vessels, \nmuscles, and nerves. Excisional surgery is considered the \npreferred treatment of choice, for which we have included \nan accompanying video.\nREFERENCES\n1. Andres MP, Arcoverde FVL, Souza CCC, Fernandes \nLFC, Abrão MS, Kho RM. Extrapelvic endometriosis: \nA systematic review. J Minim Invasive Gynecol \n2020;27(2):373–89.\n2. Wong WSF, Lim CED, Luo X. Inguinal endometriosis: \nAn uncommon differential diagnosis as an inguinal \ntumour. ISRN Obstet Gynecol 2011;2011:272159.\n3. Candiani GB, Vercellini P, Fedele L, Vendola N, \nCarinelli S, Scaglione V. Inguinal endometriosis: \nPathogenetic and clinical implications. Obstet \nGynecol 1991;78(2):191–4.\n4. Arakawa T, Hirata T, Koga K, et al. Clinical aspects \nand management of inguinal endometriosis: A \ncase series of 20 patients. J Obstet Gynaecol Res \n2019;45(10):2029–36.\n5. Niitsu H, Tsumura H, Kanehiro T, Yamaoka H, \nTaogoshi H, Murao N. Clinical characteristics \nand surgical treatment for inguinal endometriosis \nin young women of reproductive age. Dig Surg \n2019;36(2):166–72.\n6. Singh KK, Lessells AM, Adam DJ, et al. Presentation \nof endometriosis to general surgeons: A 10-year \nexperience. Br J Surg 1995;82(10):1349–51.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 9, Issue 1, 2023; Pages 11–16. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2023;9(1):11–16. \nwww.ijcriog.com\nBeroukhim et al. 15\n7. Morales Martínez C, Tejuca Somoano S. Abdominal \nwall endometriosis. Am J Obstet Gynecol \n2017;217(6):701–2.\n8. Quagliarello J, Coppa G, Bigelow B. Isolated \nendometriosis in an inguinal hernia. Am J Obstet \nGynecol 1985;152(6 Pt 1):688–9.\n9. Yang DM, Kim HC, Kim SW, Won KY. Groin \nabnormalities: Ultrasonographic and clinical findings. \nUltrasonography 2020;39(2):166–77.\n10. Gui B, Valentini AL, Ninivaggi V, Marino M, Iacobucci \nM, Bonomo L. Deep pelvic endometriosis: Don’t \nforget round ligaments. Review of anatomy, clinical \ncharacteristics, and MR imaging features. Abdom \nImaging 2014;39(3):622–32.\n11. Fedele L, Bianchi S, Frontino G, Zanconato G, Rubino \nT. Radical excision of inguinal endometriosis. Obstet \nGynecol 2007;110(2 Pt 2):530–3.\n12. Chamié LP, Ribeiro DMFR, Tiferes DA, de Macedo \nNeto AC, Serafini PC. Atypical sites of deeply \ninfiltrative endometriosis: Clinical characteristics and \nimaging findings. Radiographics 2018;38(1):309–28.\n13. Goh JT, Flynn V. Inguinal endometriosis. Aust N Z J \nObstet Gynaecol 1994;34(1):121.\n14. Nagama T, Kakudo N, Fukui M, Yamauchi T, Mitsui \nT, Kusumoto K. Heterotopic endometriosis in the \ninguinal region: A case report and literature review. \nEplasty 2019;19:ic19.\n15. Mu B, Zhang Z, Liu C, et al. Long term follow-up \nof inguinal endometriosis. BMC Womens Health \n2021;21(1):90.\n*********\nAuthor Contributions\nGabriela Beroukhim – Conception of the work, Design \nof the work, Acquisition of data, Analysis of data, \nInterpretation of data, Drafting the work, Revising the \nwork critically for important intellectual content, Final \napproval of the version to be published, Agree to be \naccountable for all aspects of the work in ensuring that \nquestions related to the accuracy or integrity of any part \nof the work are appropriately investigated and resolved\nEcem Esencan – 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\nPadmini Manrai – 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\nMasoud Azodi – Conception of the work, Design of the \nwork, Analysis of data, Interpretation of data, Revising \nthe work critically for important intellectual content, \nFinal approval of the version to be published, Agree to be \naccountable for all aspects of the work in ensuring that \nquestions related to the accuracy or integrity of any part \nof the work are appropriately investigated and resolved\nYonghee K Cho – Conception of the work, Design of the \nwork, Acquisition of data, Analysis of data, Interpretation \nof data, 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\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© 2023 Gabriela Beroukhim et al. This article is \ndistributed under the terms of Creative Commons \nAttribution License which permits unrestricted use, \ndistribution and reproduction in any medium provided \nthe original author(s) and original publisher are properly \ncredited. Please see the copyright policy on the journal \nwebsite for more information.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 9, Issue 1, 2023; Pages 11–16. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2023;9(1):11–16. \nwww.ijcriog.com\nBeroukhim et al. 16\nAccess full text article on\nother devices\nAccess PDF of article on\nother devices\n\nSubmit your manuscripts at\nwww.edoriumjournals.com","source_license":"CC0","license_restricted":false}