{"paper_id":"5f80cddb-6f2b-4269-a7b0-fae5976f333a","body_text":"Mu et al. BMC Women’s Health           (2021) 21:90  \nhttps://doi.org/10.1186/s12905-021-01235-2\nCASE REPORT\nLong term follow-up of inguinal \nendometriosis\nBoRan Mu1, ZhiQiang Zhang1, Chongdong Liu1, Kunning Zhang2, ShuHong Li1, JinHua Leng3* \nand MengHui Li1* \nAbstract \nBackground: Inguinal endometriosis (IEM) is a rare extra pelvic endometriosis. Here, we study the clinical characteris-\ntics, management strategies, and long-term gynecological outcomes of IEM patients at Beijing Chaoyang Hospital.\nCase presentation: Three patients presented with a total of four lesions (one on the left side, one on the right side, \nand one bilaterally). The diameters of the four lesions were 2 cm, 2 cm, 3.5 cm and 1.5 cm, respectively. Two patients \nwere admitted with inguinal hernias. Two patients were admitted with endometrioses—one with ovarian endometri-\nosis and one with pelvic endometriosis. The hernia sac was repaired concomitantly via excision of the round ligament \nin two patients. One patient underwent a concomitant laparoscopy for gynecologic evaluations, including an ablation \nto the peritoneal endometriosis, and resection of the left uterosacral ligament endometriosis and pelvic adhesiolysis. \nAll lesions were located on the extraperitoneal portion of the round ligament and were diagnosed histologically. No \nrecurrence was observed in the inguinal region. All patients diagnosed with adenomyosis were treated with medica-\ntion alone without any complaints.\nConclusions: Inguinal endometriosis can occur simultaneously with pelvic endometriosis. In most cases, a concomi-\ntant hernia sac appears together with groin endometriosis. Clinical management should be individualized and per-\nformed in tandem with general practitioners and obstetrics & gynecology experts. Pelvic disease, in particular, should \nbe followed-up by a gynecologist.\nKeywords: Endometriosis, Inguinal endometriosis, Hernia, Follow up, Gynecological results\n© The Author(s) 2021. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which \npermits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the \noriginal author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or \nother third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line \nto the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory \nregulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this \nlicence, visit http://creat iveco mmons .org/licen ses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creat iveco \nmmons .org/publi cdoma in/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.\nBackground\nAs a rare extra pelvic endometriosis, Inguinal endome -\ntriosis (IEM) has been reported in 0.3–0.6% of endome -\ntriosis patients [1]. IEM is also a possible site of deep \nendometriosis [2].\nAs case reports of IEM increase, so does the inci -\ndence of IEM [3]. These studies outline the clinical \ncharacteristics and the optimal diagnostic and therapeu -\ntic strategies for treating IEM [4]. However, long term \nrecovery and gynecological follow-up from IEM patients \nremains unknown for both gynecologists and general \nsurgeons.\nHere, we review cases from 3 patients with IEM who \nwere treated in our hospital. We are the first to report the \ngynecological results from long-term follow-ups in IEM \npatients.\nCase presentation\nWe identified three patients who were admitted to \nour hospital between 2009 and 2014 with pathologi -\ncally proven IEM using data from stromal cells within \nthe endometrial glands of the connective tissue in the \nOpen Access\n*Correspondence:  lengjenny@126.com; onlyjiaozi@126.com\n1 Department of Obstetrics and Gynecology, Beijing Chao-Yang Hospital \nAffiliated To Capital Medical University, 8 Gongtinanlu, ChaoYang District, \nBeijing 100020, People’s Republic of China\n3 Department of Obstetrics and Gynecology, Peking Union Medical \nCollege (PUMC) Hospital, No. 1 Shuaifuyuan Wangfujing, Dongcheng \nDistrict, Beijing 100730, People’s Republic of China\nFull list of author information is available at the end of the article\n\nPage 2 of 6Mu et al. BMC Women’s Health           (2021) 21:90 \ninguinal lump (Fig. 1A and B). The clinical characteristics \nof these cases are summarized in Table  1. Institutional \nreview board (IRB) approval was provided.\nThe durations from complaint to diagnosis were \n14 months, 6 years, 0.5 years, and 2 years, respectively, for \neach lesion. Cyclic discomfort in the inguinal region and \nconcomitance with the menstrual period was reported \nin 2 patients, while 1 complained of dysmenorrhea. Two \nlesions were reported to change in size during strenu -\nous events such as coughing. One patient previously \nunderwent a right ovarian cystectomy to address endo -\nmetriosis and infertility 15  months prior. Pre-operative \nmagnetic resonance imaging (MRI) in 2 patients detected \na solid, irregular lesion with a hypointense signal and \nsmall hemorrhagic foci with hyperintense signals using \nT1-weighted imaging in the right inguinal area (Fig.  2A, \nB). 2 patients consulted with a gynecologist and 1 patient \nwas seen by a general surgeon initially. Pre-operative \nCA125 levels were normal in 1 patient, at 25.6U/ml, and \nelevated in another, at 78.48 (normal range, 0–35) U/\nml. 2 patients were tentatively diagnosed with inguinal \n(round ligament) endometriosis.\nWe assessed 3 patients with a collective total of 4 \nlesions (1 patient had a lesion on the left side, 1 had a \nlesion on the right side, and one had bilateral lesions). \nThe diameters of the lesions were 2  cm, 2  cm, 3.5 and \n1.5  cm, respectively. 2 patients were diagnosed with \ninguinal hernias, 2 were diagnosed with endometriosis, \none was diagnosed with ovarian endometriosis, and 1 \nwas diagnosed with pelvic endometriosis. Two type III \n(Case1, left lesion of Case3) lesions adhered to the extra -\nperitoneal portion of the round ligament (Fig. 3A, B).\nTwo patients underwent procedures to remove the \nlump and repair the hernia sac. One patient had a lapa -\nroscopic ovarian cystectomy in another hospital, and \nthen underwent a procedure 15 months later to remove \nthe lump and repair the hernia. One patient underwent \nlaparoscopy for gynecologic evaluations, including a \nperitoneal endometriosis ablation, a left uterosacral liga -\nment endometriosis resection and a pelvic adhesiolysis. \nThe patient was discharged two days after the opera -\ntion. All lesions were diagnosed histologically (Fig.  4) as \nER and PR positive in the glandular and stroma (Fig.  4), \nand CD10 positive in the stroma (Fig.  4). The patients \nreceived regular follow-up evaluations and no recurrent \nlesions were observed. All patients developed adenomyo -\nsis, which was treated medicinally and followed-up in our \ndepartment.\nDiscussion\nInguinal endometriosis, a rare form of extra-genital \nendometriosis, often coincides with pelvic endome -\ntriosis. However, a concomitant hernia sac with groin \nendometriosis should also be considered in the context \nof inguinal endometriosis. Comprehensive evaluation of \npatient medical histories should be performed in tan -\ndem with imaging and individualized clinical manage -\nment strategies for IEM patients. Patients who present \nwith both pelvic and inguinal symptoms and are surgical \ncandidates for both procedures should undergo both pro-\ncedures concomitantly through collaboration between \nboth general surgery and gynecology. Follow-up evalua -\ntions should be specifically completed by a gynecologist \nto check for pelvic disease.\nThree clinical types of IEM are reported depending on \nthe site of the lesion: type 1 lesions are located at a her -\nnia sac or hydrocele of Nuck’s canal, type II lesions are \non the round ligament, and type III lesions are located \nunder the skin [4]. Type III lesions have been associated \nFig.1 Histopathological examination comprising an endometrial \nglandular structure lined by columnar epithelium, surrounded by \nendometrial-type stroma with dense fibrosis; (hematoxylin–eosin, \noriginal magnification: A ×100;     B, ×200)\n\nPage 3 of 6\nMu et al. BMC Women’s Health           (2021) 21:90 \n \nwith the hernia sac, which is an observation that differs \nbetween studies [5, 6]. Two of the 3 patients and 2 of the \n4 lesions in our report presented with concomitant her -\nnia sacs in the groin. Inguinal endometriosis often pre -\nsents concomitantly with hernia sacs in the groin region \n[7]. Understanding this characteristic could be helpful to \neffectively direct therapeutic strategies.\nUltrasonography is the first-line diagnostic method \nfor inguinal endometrioses and is used to identify con -\ncomitant hernia sacs. However the presentation of ingui -\nnal endometriosis in ultrasound is variable, including \nsolid masses, cystic masses, and combined cystic and \nsolid masses [8]. MRI is particularly useful in diagnos -\ning lesions in the extraperitoneal area, and can also be \nused to identify sub-peritoneal endometriotic deposits \n[9]. MRI scans of IEM have distinct characteristics [2], \nincluding hyperintense T1-weighted images of hemor -\nrhagic micro cysts that provide diagnostic clues for IEM \n[10].\nDifferential diagnoses of IEM include inguinal her -\nnia, hydrocele for cystic masses, sarcoma, lymphoma, \nhematoma, and abscesses for solid masses. Most IEM \npatients were initially admitted and treated by general \nsurgeons with a false diagnosis of incarcerated hernia. \nIncreased catamenial size and pain during menstrua -\ntion are hallmarks of an IEM diagnosis. The direct rela -\ntionship between symptoms and menstruation often \nsuccessfully rule out other inguinal pathologies [11]. \nHowever, surgeons should be aware of the possibility of \ninguinal endometriosis in fertile women with a lump in \nthe groin region [6 ].\nSurgery involves en bloc radical excision of the lesion \nalong with the extraperitoneal portion of the round lig -\nament [12]. A careful gynecological assessment should \nbe conducted during surgery given that intraperitoneal \nlocalization is observed in the majority of cases (91%) \n[13]. Minimally invasive surgery is the gold standard \ndiagnostic technique for identifying endometriosis \nTable 1 Characteristics of three patients with inguinal endometriosis\nCase 1 Case 2 Case 3\nAge at diagnosis 32 40 36\nGravidity and parity 2/1 0/0 3/0\nPresenting symptoms Right inguinal mass with catamenial \npain for 14mon\nLeft inguinal incarcerated mass 2y Left inguinal mass with catamenial \npain 6y and right inguinal incarcer-\nated mass 0.5y\nHistory of surgery Appendectomy in 2004 Right ovarian cystectomy in March, \n2011; Infertility\nInduced abortion 3 times; dysmenor-\nrhea\nPhysical findings Tender 2 cm nodule Tender 2 cm nodule Tender 3 cm left inguinal mass, 1.5 cm \nright inguinal nodule; uterine \nleiomyoma\nMRI findings isointense and small scattered \nhyperintense both on T1- and \nT2-weighted images\nNA Left: hyperintense both on T1- and \nT2-weighted images\nRight: hypointense on T1- and \nT2-weighted images\nType III I Left: III\nRight: I\nTentative diagnosis Pelvic and inguinal endometriosis Incarcerated hernia Inguinal endometriosis\nOperative date 2014-2-14 2012-8-14 2011-11-28\nSurgical diagnosis and treatment Endometriosis of right round liga-\nment excision and pelvic endome-\ntriosis ablation\nLeft round ligament endometriosis \nexcision; repaired the hernia;\nBilateral round ligament endometrio-\nsis excision; Hernia sac was found in \nthe right groin and was repaired\nFollow-up 70mon\nAdenomyosis; no recurrence\n88mon\nAdenomyosis; no recurrence\n96mon\nAdenomyosis; no recurrence\n\nPage 4 of 6Mu et al. BMC Women’s Health           (2021) 21:90 \n[13– 16]. Laparoscopy allows for the direct visualization \nof implants and nodules and aids in excising implants, \namplifying minimal lesions, obtaining tissue for diag -\nnosis and stage determination, and treating the disease \nappropriately.\nHormonal treatment has been underreported as a \ntherapeutic strategy for inguinal endometriosis [14]. It \ncan be an option if the patient does not want to undergo \nsurgery or does not want reoperation after recurrence, \nand it also could be indicated in patients with con -\ncomitant pelvic endometriosis [17]. Arakawa et  al. [1 ] \nreported that Dienogest effectively managed pain in \npatients who did not want surgery or reoperation after \ndisease recurrence. The expression of estrogen recep -\ntors and progesterone receptors furthers the basis for \nusing hormonal therapies for inguinal endometriosis.\nConclusions\nLong-term follow-up data regarding IEM is limited to \na few patients, and operative charts are often missing. \nDue to its rarity, IEM often lacks thorough investigation. \nThis study provides data from long term follow-ups with \nIEM patients and provides a deeper understanding of \nIEM treatment. Follow-up evaluations should continue \nto be completed by a gynecologist to monitor for intra-\nabdominal disease and to inform patients of its impact on \nfertility.\nFig.2 The magnetic resonance imagining reveals an inguinal mass, \nisointense with muscle, which infiltrate the edge (arrow) of the \nabdominis rectus muscle, in Axial T1-weighted imagine (A). Axial \nT2-weighted image, obtained at the same level (B)\n Fig.3 The mass is freed from the adhesions with the internal oblique \nmuscle and the transversalis fascia at the deep inguinal orifice. The \ninguinal segment of the round ligament was excised with the lesion \n(A). Multi-locular cysts containing dark hemorrhagic content was \nrevealed by gross specimen (B)\n\nPage 5 of 6\nMu et al. BMC Women’s Health           (2021) 21:90 \n \nAbbreviations\nIEM: Inguinal endometriosis; MRI: Magnetic resonance imaging; ER: Estrogen \nreceptor; PR: Progesterone receptor.\nAcknowledgements\nNot applicable.\nAuthors’ contributions\nMHL contributed to the design of the study, data collection and analysis, inter-\npretation of the analyses, writing and revising the manuscript. JHL contributed \nto the analysis, writing, and revising of the manuscript. ZhQZh and BRM \ncontributed substantially to the study’s conception and design, drafted the \narticle, and revised the approved final version to be published. ChDL, KNZh \nand ShHL contributed to the design of the study and revising the manuscript. \nAll authors approved the final version of the manuscript and agreed to be \nresponsible for all aspects of the work.\nFunding\nThis work was supported by funding from the National Key R&D Program of \nChina Number: 2017YFC1001204.\nAvailability of data and materials\nAll data generated or analyzed during this study are included in this published \narticle.\nDeclarations\nEthical approval and consent to participate\nThis study protocol was approved by the Institutional Review Board (IRB) of \nBeijing Chao-Yang Hospital affiliated to Capital Medical University (IRB no. \n2016-science-166). All patients received information on the purpose and \nprocedures of this study, and provided written, informed consent.\nFig.4 Focal endometriosis, which consisted of endometrial gland and stroma inside the tissue (H&E ×200) of case 1, 2, 3 respectively. ER and PR \nwere positive in glandular and stroma, and CD10 was positive in stroma (× 200)\n\nPage 6 of 6Mu et al. BMC Women’s Health           (2021) 21:90 \n•\n \nfast, convenient online submission\n •\n  \nthorough peer review by experienced researchers in your field\n• \n \nrapid publication on acceptance\n• \n \nsupport for research data, including large and complex data types\n•\n  \ngold Open Access which fosters wider collaboration and increased citations \n \nmaximum visibility for your research: over 100M website views per year •\n  At BMC, research is always in progress.\nLearn more biomedcentral.com/submissions\nReady to submit y our researc hReady to submit y our researc h  ?  Choose BMC and benefit fr om: ?  Choose BMC and benefit fr om: \nConsent for publication\nWritten informed consent was acquired from each patient for this publica-\ntion using a BMC consent form and is available for review by the editor of this \njournal.\nCompeting interests\nThe author(s) declared no potential conflicts of interest with the research, \nauthorship, and/or publication of this article.\nAuthor details\n1 Department of Obstetrics and Gynecology, Beijing Chao-Yang Hospital \nAffiliated To Capital Medical University, 8 Gongtinanlu, ChaoYang District, \nBeijing 100020, People’s Republic of China. 2 Department of Pathology, Beijing \nChao-Yang Hospital Affiliated to Capital Medical University, 8 Gongtinanlu, \nChaoYang District, Beijing 100020, People’s Republic of China. 3 Department \nof Obstetrics and Gynecology, Peking Union Medical College (PUMC) Hospital, \nNo. 1 Shuaifuyuan Wangfujing, Dongcheng District, Beijing 100730, People’s \nRepublic of China. \nReceived: 10 September 2020   Accepted: 22 February 2021\nReferences\n 1. 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Obstet \nGynecol Sci. 2014;57(2):172–5.\nPublisher’s Note\nSpringer Nature remains neutral with regard to jurisdictional claims in pub-\nlished maps and institutional affiliations.","source_license":"CC0","license_restricted":false}