{"paper_id":"89ba975c-c0b1-429f-b0c8-59a19710e88d","body_text":"Journal of Case Reports and Images in Obstetrics and Gynecology, Volume 11, Issue 1, 2025; Pages 42–47. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2025;11(1):42–47.   \nwww.ijcriog.com\nDaniels et al. 42\nCASE REPORT  PEER REVIEWED | OPEN ACCESS\nCase of deep infiltrating and cystic endometriosis in a  \npatient with a Müllerian anomaly: Surgical treatment by \nremoving non-communicating horn\nTanne Daniels, Kaat Moelans, Jasper Verguts, Jurgen Bielen\nABSTRACT\nIntroduction: Endometriosis has a higher incidence in \npatients with Müllerian anomalies due to the increased \nlikelihood of retrograde menstruation. This case presents \nunique challenges due to the diverse morphologies seen \nin Müllerian anomalies. Therefore, accurate and timely \npreoperative diagnosis is essential to optimize pain \nmanagement and fertility outcomes.\nCase Report:  This case report presents the case of \na 31-year-old patient with grade 4 endometriosis and \na Müllerian anomaly, which resulted in the surgical \ntreatment of the non-communication horn as well as the \ndiseased ovary.\nConclusion: This case exemplifies the challenges seen \nin with Müllerian anomaly-associated endometriosis, \nsuch as diagnostic delay and intricacy of surgical \ntreatment options. This case highlights the importance \nof early intervention and a multidisciplinary approach. \nIncreased awareness of endometriosis in patients with \nMüllerian anomalies among healthcare providers can \nimprove outcomes and minimize complications.\nKeywords: Cystic endometriosis, Endometriosis, Horn \nexcision, Müllerian anomaly\nTanne Daniels1, Kaat Moelans2, Jasper Verguts3, Jurgen Bielen4\nAffiliations: 1Fellow Benign Gynecology and Endometriosis, \nObstetrics and Gynecology, Jessa Hospital Hasselt, Hasselt, \nBelgium; 2Medical student, Obstetrics and Gynecology, Jessa \nHospital Hasselt, Hasselt, Belgium; 3Gynecologist, Obstetrics \nand Gynecology, Jessa Hospital Hasselt, Hasselt, Belgium; \n4Radiologist, Radiology Department, Jessa Hospital Hasselt, \nHasselt, Belgium.\nCorresponding Author: Tanne Daniels, Fellow Benign Gy -\nnecology and Endometriosis, Obstetrics and Gynecology, \nJessa Hospital Hasselt, Hasselt, Belgium; Email: tannedan-\niels@hotmail.com\nReceived: 04 January 2025\nAccepted: 12 February 2025\nPublished: 08 March 2025\nHow to cite this article\nDaniels T, Moelans K, Verguts J, Bielen J. Case \nof deep infiltrating and cystic endometriosis in a  \npatient with a Müllerian anomaly: Surgical treatment by \nremoving non-communicating horn. J Case Rep Images \nObstet Gynecol 2025;11(1):42–47.\nArticle ID: 100197Z08TD2025\n*********\ndoi: 10.5348/100197Z08TD2025CR\nINTRODUCTION\nEndometriosis is a common gynecological condition \ncharacterized by benign proliferation of endometrium-\nlike tissue outside of the uterine cavity [1–3]. This ectopic \ntissue can be found on the ovaries, fallopian tubes, outer \nsurface of the uterus, and other organs within the pelvis. \nUnlike the endometrial tissue within the uterus, which is \nshed during menstruation, this ectopic tissue can cause \ninflammation, scarring, and adhesions. This can lead to \nchronic pain, heavy periods, and fertility issues. The exact \ncause of endometriosis is not well understood, but it is \nbelieved to involve a combination of genetic, hormonal, \nimmune, and environmental factors [4, 5].\nRetrograde menstruation is thought to play a key role \nin the disease development [3, 4]. This retrograde flow of \nmenstrual fluids facilitates spreading of endometrial tissue \ninto the abdominal cavity. Retrograde menstruation can \noccur in healthy individuals but is more frequent in women \nwith Müllerian anomalies. These uterine malformations \noccur in 0.2–5% of the female population and can result \nin obstructive or preserved outflow tracts [6]. However, \nthis is not the only factor to be involved in the disease \ndevelopment of endometriosis. Pathophysiology believes \nthat the disease is estrogen-dependent with the presence \nof estrogen receptors found in endometriosis tissue as \nwell as an inflammatory disease [4].\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 11, Issue 1, 2025; Pages 42–47. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2025;11(1):42–47.   \nwww.ijcriog.com\nDaniels et al. 43\nThe patient presented in this report was diagnosed \nwith a unicornuate uterus and a left-sided rudimentary \nhorn. Unicornuate uteruses occur in approximately \n1 in 1000 to 1 in 5400 women, of which 74–90% have \nan associated rudimentary horn [7]. These patients \nare susceptible to gynecologic complications including \npremature birth and miscarriage, hematosalpinx, chronic \npelvic pain, and endometriosis [8].\nRetrospective studies showed high endometriosis \nrates in women with Müllerian anomalies. Due to \nretrograde menstruation, the correlation is significantly \nhigher in case of obstructive uterine malformations. The \npresence of endometrium tissue in the rudimentary horn \nis another important factor. Endometriosis occurrence is \nconsiderably higher in patients with active endometrium in \nthe non-communicating horn, compared to those without \nactive endometrium. In addition, uterine malformations \nmight also lead to impaired uterine peristalsis, causing \nretrograde flow regardless of outflow tract function [6, 9, \n10]. In conclusion, the correlation between obstructive \nMüllerian anomalies and endometriosis might be \nexplained by higher rates of retrograde menstruation.\nThe diagnosis of rudimentary horns is often delayed, \ndue to the presence of normal menstruation from the \nunicornuate uterus, absent clinical manifestation, and \ndifficult visualization on imaging. The gold standard for \nvisualization of rudimentary uterine horns is magnetic \nresonance imaging (MRI). Importantly, MRI is helpful \nin determining whether the rudimentary horn contains \nfunctional endometrium. After diagnosis, resection of the \nrudimentary horn is the recommended treatment. This \nis thought to prevent further complications and improve \nfertility outcomes [7, 11–13].\nWe discuss the surgical management of this case, \nwhich included removal of the rudimentary horn and \nextensive endometriosis lesions.\nCASE REPORT\nWe present a 31-year-old female who was evaluated \nat a primary care center following a two-week history \nof acute-onset lower abdominal pain. Initial imaging by \npelvic ultrasound showed an endometrioma measuring \n5 cm. Upon this finding, she was urgently referred to \nour center and began treatment with a combination \ncontraceptive pill containing ethinyl estradiol and \ndienogest. She was seen in clinic two days later, with \ncomplaints including grade 3 dysmenorrhea, progressive \nlower abdominal pain, dyschezia, and progressive \nfatigue. The pain escalated in severity, leading to \nsymptoms of nausea and dizziness, necessitating the use \nof multiple analgesics for relief (including tramadol). \nOn clinical examination, the speculum assessment was \nunremarkable, with no nodules observed. Although \nvaginal palpation elicited pain, the overall findings were \nnormal. On pelvic ultrasound scan a bicorn uterus was \nsuspected with normal trilaminar endometrium in the \nright horn and an anechogenic intrauterine lesion with \nhyperechogenic borders. A homogenic ground glass \ncyst was observed between both uterine horns. The cyst \nseemed fixed against the uterus and measured 6 cm in \nmaximum diameter with a color score of 1. These findings \nwere compatible with an endometrioma, originating \nfrom the left ovary. The right ovary was morphologically \nnormal, but less mobile against the uterus. The anterior \ncompartment appeared normal on ultrasound scan. The \nposterior compartment had a thickened left sacro-uterine \nligament, without obvious deep infiltrating nodule.\nThe patient’s medical history mentions a bicornuate \nuterus and a right-sided renal agenesis. Her gynecological \nhistory shows gravida 1, para 1, with a spontaneous \nconception after 1.5 years. The baby was delivered \nthrough cesarian section because of breech position. \nUpon presentation in our clinic, she expressed a persisting \npregnancy wish.\nFurther investigations showed an anti- Müllerian \nhormone (AMH) value of 2.62 µg/L. An MRI scan was \nconducted as echographic evaluation of the posterior \ncompartment was not optimal in the presence of the \nendometrioma and because of the higher probability of a \ndeep infiltrating endometriosis lesions in the presence of \nan endometrioma.\nTwo weeks after the consultation in our center the \npatient referred to the emergency department because \nof acute on chronic pain. An abdominal ultrasound and \ncomputed tomography (CT) scan showed peritonitis, \nlikely secondary to appendicitis. Upon these findings, \nurgent laparoscopic abdominal surgery was performed. \nHere, an inflamed appendix was observed, along with \nchocolate-brown fluid in the pelvic region (consistent with \nthe rupture of an endometrioma). An appendectomy was \nperformed and the pelvis was washed out. Histologically, \nendometriosis of the appendix was found, without signs \nof appendicitis.\nThe MRI scan showed a unicornuate uterus with \ndilatation of the hypotrophic, non-communicating left \nhorn with blood pooling (Figure 1A and B). It also showed \na right tubo-ovarian complex with a likely endometrioma \nwith recent blood. On the left side a large tubo-ovarian \ncomplex with hematosalpinx with large endometrioma \n(5.5 cm) and encapsulated serosanguinous fluid adjacent \nto the ovary could be seen. The sacro-uterine ligaments \nwere bilaterally thickened with underlying cysts right \nparamedian, extending to the adjacent wall of the distal \nsigmoid with no deep bowel infiltration (Figure 2).\nThe case, together with MRI findings, was discussed at \nthe multidisciplinary endometriosis meeting. This team \nof medical experts consists of gynecologists, radiologists, \nabdominal surgeons, urologists, as well as sexologists \nand fertility doctors. We decided on treatment by surgical \nremoval of the left rudimentary horn along with the \nexcision of endometriosis lesions. The distance between \nthe two horns appeared sufficient to safely remove the \nleft horn. We planned for preoperative stenting of the \nureter since she had a single kidney. Due to the extent \nof the disease, the surgery was planned to be performed \nrobotically.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 11, Issue 1, 2025; Pages 42–47. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2025;11(1):42–47.   \nwww.ijcriog.com\nDaniels et al. 44\nPerioperatively, we found a large tubo-ovarian complex \n(endometrioma with large hematosalpinx) on the left \nside, adjacent to the ovarian fossa. The right horn of the \nuterus appeared normal, as well as the right tubo-ovarian \ncomplex, apart from superficial ovarian endometriosis \nlesions. The rectosigmoid was folded over this complex \nand the right horn of the uterus. The upper abdomen \nappeared to be free of endometriosis lesions. At the start \nof the surgery the non-communicating left horn was \nnot visible. We began with extensive adhesiolysis of the \nrectosigmoid, which proceeded without complications. \nAfter the adhesiolysis the non-communicating left horn \nbecame visible.\nWe proceeded to remove the left tubo-ovarian \ncomplex, as there was no visible normal ovarian tissue \nremaining. If diagnosed earlier there would have been a \npossibility to save the ovary; however, this is hard to say. \nFollowing this, the rudimentary left horn was excised. \nAdditionally, a recto-vaginal nodule without bowel or \nvaginal infiltration was removed, resulting in a stage \n4 AFS endometriosis level. The left lateral incision was \nbroadened to remove the specimen. The ureter stent was \nremoved during the same operation.\nThe immediate postoperative recovery was \nuncomplicated, and the patient was able to leave the \nhospital the next day. She was advised to use a hormonal \ncontraceptive until three months postoperatively. After \nthis period, she may try to conceive again.\nAt the six-week postoperative follow-up consultation, \nshe showed favorable recovery, with satisfactory healing \nof incision sites and no signs of recurrence on transvaginal \nultrasound. Because of her persistent pregnancy wish, she \ndecided against using contraceptives after the procedure. \nLuckily, an active cycle with a dominant follicle on the \nright ovary were established on ultrasound imaging.\nDISCUSSION\nAccording to the literature, approximately 75% \nof women with a uterine malformation also have \nendometriosis [6, 14], which is significantly higher than \nthe estimated 10% prevalence in the general population \n[15–17]. Additionally, the association between uterine \nmalformations and infertility may exacerbate the severity \nof endometriosis, highlighting the importance of accurate \ndiagnosis and effective management [9].\nIn this case the diagnosis of a Müllerian anomaly \nwas made before the patient’s first pregnancy, but no \nlink was made with the possibility of the presence of \nendometriosis. She reported to the general practitioner \nand gynecologist multiple times with complaints of \ndysmenorrhea and lower abdominal pain, but no \ndiagnosis was made. She was finally diagnosed after the \npresence of a large endometrioma was seen following an \nacute on chronic abdominal pain episode for which she \npresented to the emergency department. This resulted \nin a diagnostic delay for this patient of about five years, \nwhich is consistent with literature [18].\nThe increased prevalence of endometriosis in \npatients with Müllerian anomalies is well documented. \nAdditionally, ipsilateral renal agenesis has been \nassociated with Müllerian anomalies and, consequently, \nendometriosis [19].\nAs retrograde menstruation plays a big role in the \npathogenesis of endometriosis in these patients, early \ndiagnosis and surgical management are crucial for the \nprevention of endometriosis lesions [6, 8, 9, 14, 19].\nIn the latest systemic review by Abboud et al. it is \nstated that laparoscopic removal of the rudimentary horn \nshould be considered as a therapeutic option [11]. This \ncase report underscores the importance of surgically \nremoving the rudimentary horn in treating endometriosis \nwhen symptoms arise from retrograde menstruation. \nWithout this intervention, the disease is more likely to \nrecur.\nWith this case report we want to underline the \nimportance of correct preoperative imaging, including \nMRI, for the specifications according the Müllerian \nanomaly. Furthermore, a multidisciplinary approach \nshould be favored to thoroughly estimate the best \npossible surgical treatment. In patients with known \nMüllerian anomalies physicians should be vigilant of the \npossibility of the presence of endometriosis, especially \nwith subsequent complaints.\nFigure 1: (A and B) T2 and T1 MRI image of the congenital \nanomaly with a unicornuate uterus (right cavity normal) and a \nhypoplastic, non-communicating left horn; dilation of the left \nhorn containing blood. Discrete hemorrhagic endometriosis \nfoci adjacent to the serosa on the left.\nFigure 2: T2 MRI image of the two horns of the uterus with the \nleft ovarium endometrioma in between.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 11, Issue 1, 2025; Pages 42–47. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2025;11(1):42–47.   \nwww.ijcriog.com\nDaniels et al. 45\nCONCLUSION\nThis case report exemplifies the challenges seen in \nwith Müllerian anomaly-associated endometriosis, such \nas diagnostic delay and intricacy of surgical treatment \noptions. This case highlights the importance of early \nintervention and a multidisciplinary approach. Increased \nawareness of endometriosis in patients with Müllerian \nanomalies among healthcare providers can improve \noutcomes and minimize complications.\nREFERENCES\n1. Patel BG, Lenk EE, Lebovic DI, Shu Y, Yu J, Taylor \nRN. Pathogenesis of endometriosis: Interaction \nbetween endocrine and inflammatory pathways. 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Cham: Springer \nInternational Publishing; 2016. p. 119–32.\n8. Pitot MA, Bookwalter CA, Dudiak KM. Müllerian duct \nanomalies coincident with endometriosis: A review. \nAbdom Radiol (NY) 2020;45(6):1723–40.\n9. Boujenah J, Salakos E, Pinto M, et al. Endometriosis \nand uterine malformations: Infertility may increase \nseverity of endometriosis. Acta Obstet Gynecol Scand \n2017;96(6):702–6.\n10. Freytag D, Mettler L, Maass N, Günther V, Alkatout I. \nUterine anomalies and endometriosis. Minerva Med \n2020;111(1):33–49.\n11. Abboud K, Giannini A, D’Oria O, et al. Laparoscopic \nmanagement of rudimentary uterine horns in patients \nwith unicornuate uterus: A systematic review. Gynecol \nObstet Invest 2023;88(1):1–10.\n12. Shukunami K, Tsunezawa W, Kotsuji F. Unicornuate \nuterus with a noncommunicating cavitary, laterally \ndislocated rudimentary horn presenting with \nadenomyosis, associated with ipsilateral renal \nagenesis. Arch Gynecol Obstet 2000;264(2):88–9.\n13. Osnytska Y, Stojko R, Drosdzol-Cop A. The 18-year-\nold-girl with unicornuate uterus and endometriosis. \nGinekol Pol 2024;95(2):167–8.\n14.\t Kapczuk\t K,\tFriebe\tZ,\tIwaniec\tK,\tKędzia\tW.\tObstructive\t\nMüllerian anomalies in menstruating adolescent \ngirls: A report of 22 cases. J Pediatr Adolesc Gynecol \n2018;31(3):252–7.\n15. Becker CM, Bokor A, Heikinheimo O, et al. ESHRE \nguideline: Endometriosis. Hum Reprod Open \n2022;2022(2):hoac009.\n16. Kuznetsov L, Dworzynski K, Davies M, Overton C; \nGuideline Committee. Diagnosis and management \nof endometriosis: Summary of NICE guidance. BMJ \n2017;358:j3935.\n17. Moradi Y, Shams-Beyranvand M, Khateri S, et al. A \nsystematic review on the prevalence of endometriosis \nin women. Indian J Med Res 2021;154(3):446–54.\n18. Horne AW, Missmer SA. Pathophysiology, \ndiagnosis, and management of endometriosis. BMJ \n2022;379:e070750.\n19. Yu BR, Chae HS, Rheu CH. Early-onset severe \novarian endometriosis in adolescents with completely \nobstructed Müllerian anomalies accompanied by \nipsilateral renal agenesis: Two case reports. J Surg \nCase Rep 2024;2024(5):rjae272.\n*********\nAuthor Contributions\nTanne Daniels – Conception of the work, Design of the \nwork, Acquisition of data, Analysis of data, Interpretation \nof data, Drafting the work, Final approval of the version \nto be published, Agree to be accountable for all aspects of \nthe work in ensuring that questions related to the accuracy \nor integrity of any part of the work are appropriately \ninvestigated and resolved\nKaat Moelans – Conception of 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\nJasper Verguts – Interpretation of data, 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\nJurgen Bielen – Interpretation of data, 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\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.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 11, Issue 1, 2025; Pages 42–47. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2025;11(1):42–47.   \nwww.ijcriog.com\nDaniels et al. 46\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© 2025 Tanne Daniels et al. This article is distributed \nunder the terms of Creative Commons Attribution \nLicense which permits unrestricted use, distribution \nand reproduction in any medium provided the original \nauthor(s) and original publisher are properly credited. \nPlease see the copyright policy on the journal website for \nmore information.\nABOUT THE AUTHORS\nArticle citation: \tDaniels\tT,\tMoelans\tK,\tVerguts\tJ,\tBielen\tJ.\tCase\tof\tdeep\tinfiltrating\tand\tcystic\tendometriosis\t in\ta\t \npatient with a Müllerian anomaly: Surgical treatment by removing non-communicating horn. J Case Rep Images \nObstet Gynecol 2025;11(1):42–47.\nTanne Daniels\tis\ta\tgynecologist\twith\ta\tspecific\tinterest\tin\tendometriosis\tand\tbenign\tgynecology.\tShe\t\nearned her master’s degree in medicine and specialistic medicine (Gynecology and Obstetrics) from \nthe\t Catholic\t University\t in\t Leuven,\t Belgium.\t She\t pursued\t a\t fellowship\t in\t the\t field\t of\t endometriosis\t\nand benign gynecology at The Jessa Hospital in Hasselt, Belgium and is currently working in the \nendometriosis centre in the Jeroen Bosch in the Netherlands, where she provides care to patients \ndealing with endometriosis and collaborates on research aimed at improving treatment options. She \nhas published 3 research papers in national and international academic journals.\nEmail: Tannedaniels@hotmail.com\nJasper Verguts is a gynecologist at Jessa Hospital in Hasselt, Belgium. He earned his mater’s degree \nin medicine and specialized medicine at the Catholic University of Leuven, Belgium. Afterward he \ncontinued\twith\ta\tdoctoral\tdegree\tin\t2011\tin\tthe\tfield\tof\tendoscopic\tsurgery.\tHe\tset\tup\tthe\tendometriosis\t\ncentre in Hasselt, where he provides care to patients dealing with endometriosis and collaborates on \nresearch aimed at improving treatment options of this condition. He has published diverse research \npapers in national and international academic journals and authored 1 book in the subject of \nendometriosis.\nKaat Moelans\tis\ta\tmedical\tstudent\tat\tthe\tUniversity\tof\tGent\twith\ta\tspecific\tinterest\tin\tGynaecology\t\n& Obstetrics. She is pursuing to become a gynecologist and plans to start her residency training in \nSwitzerland after graduating.\nJurgen Bielen is a radiologist in the Radiology Department at the Jessa Hospital in Hasselt, Belgium.\nHe has a master’s degree in medicine and civil engineering and subspecialized in the imaging of the \nabdomen. He is a profound member of the Multidisciplinary Endometriosis Team at Jessa Hospital, \nwhere he continues to provide care in patients dealing with this condition.\n\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 11, Issue 1, 2025; Pages 42–47. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2025;11(1):42–47.   \nwww.ijcriog.com\nDaniels et al. 47\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}