{"paper_id":"ea0abaa2-9f49-4bb5-a209-69270c30efb7","body_text":"Intestinal Perforation due to Deep In filtrating\nEndometriosis during Pregnancy: Case Report\nRotura intestinal durante a gravidez devido a\nendometriose profunda in filtrativa: relato de caso\nMárcia Mendonça Carneiro 1 Luciana Maria Pyramo Costa 1 Maria Das Graças Torres 1\nPatrícia Salomé Gouvea 1 I v e t ed eÁ v i l a1\n1 Department of Obstetrics and Gynecology, Universidade, Federal de\nMinas Gerais, Belo Horizonte, Minas Gerais, Brazil\nRev Bras Ginecol Obstet 2018;40:235 –238.\nAddress for correspondence Márcia Mendonça Carneiro, MD, PhD,\nUniversidade Federal de Minas Gerais, Rua Antonio Torres 186,\nBelo Horizonte, MG 31030-130, Brazil\n(e-mail: marciamc.ufmg@gmail.com).\nKeywords\n► deep in filtrating\nendometriosis\n► intestinal\nendometriosis\n► intestinal rupture\n► pregnancy\ncomplications\nAbstract We report the case of a 33 year-old woman who complained of severe dysmenorrhea\nsince menarche. From 2003 to 2009, she underwent 4 laparoscopies for the treatment\nof pain associated with endometriosis. Afte r all four interventions, the pain recurred\ndespite the use of gonadotropin-releasing hormone (GnRH) analogues and the\ninsertion of a levonorgestrel intrauterine system (LNG-IUS). Finally, a colonoscopy\nperformed in 2010 revealed rectosigmoid stenosis probably due to extrinsic compres-\nsion. The patient was advised to get pregnant before treating the intestinal lesion.\nSpontaneous pregnancy occurred soon after LNG-IUS removal in 2011. In the 33rd\nweek of pregnancy, the patient started to feel severe abdominal pain. No fever or sings\nof pelviperitonitis were present, but as the pain worsened, a cesarean section was\nperformed, with the delivery of a premature healthy male, and an intestinal rupture was\nidentified. Severe peritoneal infection and sepsis ensued. A colostomy was performed,\nand the patient recovered after eight days in intensive care. Three months later, the\ncolostomy was closed, and a new LNG-IUS was inserted. The patient then came to be\ntreated by our multidisciplinary endometriosis team. The diagnostic evaluation\nrevealed the presence of intestinal lesions with extrinsic compression of the rectum.\nShe then underwent a laparoscopic excision of the endometriotic lesions, including an\novarian endometrioma, adhe siolysis and segmental cole ctomy in 2014. She is now fully\nrecovered and planning a new pregnancy. A transvaginal ultrasound (TVUS) performed\nsix months after surgery showed signs of pelvic adhesions, but no endometriotic\nlesions.\nResumo Relatamos o caso de uma mulher de 33 anos qu e apresentava de dismenorreia grave\ndesde a menarca. Entre 2003 e 2009, a paciente foi submetida a quatro laparoscopias\npara o tratamento de dor associada à endometriose. A dor persistiu apos as 4 cirurgias\napesar do uso de análogos do hormônio de liberação de gonadotropina (GnRH) e da\ninserção de um sistema intrauterino de levonorgestrel (SIU-LNG). Finalmente, uma\nreceived\nSeptember 17, 2017\naccepted\nDecember 20, 2017\nDOI https://doi.org/\n10.1055/s-0038-1624579.\nISSN 0100-7203.\nCopyright © 2018 by Thieme Revinter\nPublicações Ltda, Rio de Janeiro, Brazil\nTHIEME\nCase Report 235\n\n\nIntroduction\nEndometriosis is a progressive and benign estrogen-depen-\ndent disease de fined by the presence of endometrial tissue\n(glands and stroma) outside the uterine cavity. Deep infiltrat-\ning endometriosis (DIE) is considered a speci fic entity, which\nhas been arbitrarily de fined in histological terms as endo-\nmetriotic lesions extending more than 5 mm underneath the\nperitoneum, and it is responsible for painful symptoms, with\nsome women experiencing severe symptoms, while others\nremain asymptomatic.\n1 Due to the variable clinical presenta-\ntion, the lack of pathognomonic symptoms, and the fact that no\nuseful noninvasive clinical tests to diagnose the symptomatic\ndisease are available, a delay in the diagnosis that averages\nfrom 5 to 11 years is observed. 1,2\nDeep infiltrating endometriosis is found in 20% of women\nwith endometriosis. Bowel involvement is diagnosed in 5% to\n12% of patients with endometriosis, with most lesions (90%)\nlocated in the colorectum. The role of colorectal endometri-\nosis in women with infertility remains to be established.\n1,3\nUnfortunately, in most published studies, fertility and preg-\nnancy data are underreported or not fully considered.\n4\nIntestinal DIE is a severe disease that may affect young\nwomen desiring pregnancy. Digestive symptoms may be\nfound in association with deep dyspareunia, infertility, and\nimpaired quality of life. In order to determine the best\napproach to bowel endometriosis, several factors such as\nclinical symptoms, extent of the disease and imaging evalu-\nation results should be taken into consideration.\n2,4,5\nCase Summary\nWe report the case of a 33 year-old woman who complained of\nsevere dysmenorrhea since menarche. From 2003 to 2009, she\nunderwent 4 laparoscopies for the treatment of pain associated\nwith endometriosis (dysmenorrhea, dyschezia). The first sur-\ngery, which was performed in 2003, revealed endometriosis\ngrade IV. A gonadotropin-releasing hormone (GnRH) analogue\n(goserelin) was administered to her for 6 months after surgery,\nbut the pain recurred soon after her menses returned. As the\npelvic pain persisted, a new laparoscopy was performed in\n2006, and the levonorgestrel intrauterine system (LNG-IUS,\nMirena, Bayer, Leverkusen, Germany) was inserted, with par-\ntial pain relief. In 2008, she started complaining of dyschezia,\nand underwent a third laparoscopy, for which no records are\navailable. In spite of using Mirena, the pain, as well as the\ndyschezia, persisted. Thus, she underwent a fourth surgery in\n2009. Finally, a colonoscopy performed in 2010 revealed\nrectosigmoid stenosis probably due to extrinsic compression.\nShe was advised to get pregnant before attempting to treat the\nintestinal lesion, in view of the risks involved in such a surgery.\nSpontaneous pregnancy occurred soon after the LNG-IUS\nwas removed in 2011. When the patient was in the 33rd week\nof pregnancy, she started feeling severe abdominal pain. No\nfever or sings of pelviperitonitis were present, but as the pain\nworsened, a cesarean section was performed, with the delivery\nof a premature healthy male. Unfortunately, an intestinal\nrupture was also identi fied. Severe peritoneal infection and\nsepsis ensued. A colostomy was performed, and the patient\nrecovered after 8 days in intensive care. Three months later, the\ncolostomy was closed, and a new Mirena was inserted. Only\nthen did the patient come for treatment of the intestinal\nendometriosis with our multidisciplinary endometriosis\nteam. The diagnostic evaluation, including a transvaginal\nultrasound (TVUS) with intestinal preparation and a magnetic\nresonance imaging (MRI) scan (\n►Fig. 1), revealed the presence\nof intestinal lesions, while the colonoscopy confirmed stenosis\ndue to extrinsic compression of the rectum ( ►Fig. 2 ).\nThe patient then underwent a laparoscopic excision of the\nendometriotic lesions, including an ovarian endometrioma,\nadhesiolysis and segmental colectomy on November 2014.\nShe is now fully recovered and planning a new pregnancy.\nA TVUS performed six months after surgery showed signs of\npelvic adhesions, but no endometriotic lesions.\ncolonoscopia realizada em 2010 revelou estenose rectosigmoide, provavelmente\ndevido à compressão extrínseca. A paciente foi aconselhada a engravidar antes de\ntratar a lesão intestinal. A gravidez espontânea ocorreu logo após a remoção de LNG-\nIUS em 2011. Na 33ª semana de gestação, a paciente começou a sentir dor abdominal\nintensa, sem febre ou sinais de peritonite. Como a dor piorou consideravelmente, a\npaciente foi submetida à cesariana com nascimento prematuro de um menino\nsaudável. Durante a cesárea foi identi ficado rotura intestinal com peritonite grave e\nsepse. Uma colostomia foi realizada, e a paciente admitida no centro de terapia\nintensiva por 8 dais. A colostomia foi fechada e um novo SIU-LNG inserido. A paciente\npassou a ser tratada pela nossa equipe multid isciplinar de endometriose. A avaliação\ndiagnóstica revelou a presença de lesões in testinais com compressão extrínseca do\nreto. Foi então submetida a uma excisão laparoscópica das lesões endometrióticas,\nincluindo um endometrioma ovariano, ades iólise e colectomia segmentar em 2014. Ela\ne s t áa g o r at o t a l m e n t er e c u p e r a d aep l a n e j an o v ag r a v i d e z .U m au l t r a s s o n o g r afia\ntransvaginal (TVUS) realizada seis meses após a cirurgia revelou sinais de aderências\npélvicas sem lesões de endometriose.\nPalavras-Chave\n► endometriose\ninfiltrativa\nprofunda\n► endometriose\nintestinal\n► rotura intestinal\n► complicações na\ngravidez\nRev Bras Ginecol Obstet Vol. 40 No. 4/2018\nIntestinal Perforation due to Endometriosis during Pregnancy Carneiro et al.236\n\n\nDiscussion\nIntestinal evaluation is extremely important for the surgical\nplanning, since the number of lesions and the depth of invasion\ninfluence the composition of the surgical team, the equipment\nused and the technique chosen. Bowel involvement is frequently\nmultifocal, and the most commonly affected areas are the\nrectosigmoid colon, the appendix, the cecum and the distal\nileum.\n1,5 Although the rectal endoscopic sonographic approach\nis the most precise approach for the evaluation of the involve-\nment of intestinal layers, such identification is also possible by\nTVUS.4,5 It has been shown that lesions that affect more than\n40% of the bowel circumference reach beyond the inner mus-\ncular layer.\n3–5\nWomen presenting with intestinal symptoms and those with\nprevious endometriosis surgery are at increased risk of bowel\nresection.\n4,5 Our patient presented with recurrent symptoms,\nincluding dyschezia, despite four laparoscopies and the contin-\nuous use of medical treatments. The decision to perform surgery\nfor DIE is mainly clinical.\n4,5 The TVUS and other imaging\nt e c h n i q u e ss u c ha st h eM R Ic a nb eu s e f u lt o o l st om a k ea\npreoperative estimate of the size and lateral extension of the\nlesions, and they play a vital role in the surgical planning and\napproach. It remains unclear, however, to what extent the\npreoperative ultrasonography or the MRI should influence the\ndecision to perform the surgery, or the decision regarding\nthe type of intervention to undertake for DIE.\n5,6 According to\nt h eE u r o p e a nS o c i e t yo fH u m a nR e p r o d u c t i o na n dE m b r y o l o g y\n(ESHRE) Guideline,\n2 the ureter, the bladder and the bowels\nshould be assessed if DIE is suspected, in order to establish\nthe extent of the disease. Even though the colonoscopy per-\nformed in 2010 revealed rectosigmoid stenosis probably due to\nextrinsic compression, the patient was advised to get pregnant\nbefore attempting to treat the intestinal lesion, in view of the\nr i s k si n v o l v e di ns u c has u r g e r y .A st h ep a t i e n to n l yc a m et oo u r\nmultidisciplinary team after intestinal rupture in 2013, we do\nnot know why she was not operated earlier, even though the\ndecision to perform surgery may vary according to the literature.\nAlthough the accuracy of the colonoscopy for the identi fi-\ncation of intestinal involvement in DIE is debatable\n7 in view of\nthe rarity of mucosal involvement, it was indicated in our\npatient due to the persistent intestinal symptoms. Indeed, the\ncolonoscopy showed stenosis due to extrinsic compression.\nTherefore, surgery should have been contemplated before\npregnancy, even though there is no consensus in the medical\nliterature as to what is the best approach in such clinical\nsettings.\n3,6 Once again, she was not previously under our\ncare, and no record of clinical decisions was available to us.\nSurgery is considered the treatment of choice for symp-\ntomatic DIE, as the complete removal of lesions results in\nsignificant pain relief and improvement in quality of life\nscores.2,4,5 The best therapeutic approach for women with\nDIE involving the sigmoid and/or rectum remains to be\nestablished.4,5 A variety of factors such as clinical symptoms,\nlesion location and results from imaging studies (TVUS and\nMRI), as well as the recurrence rates and impact on fertility\nand quality of life should be taken into consideration. 5,6\nSome advocate that a bowel resection is rarely justi fied,\nFig. 1 Magnetic resonance imaging scan showing an endometriotic\nnodule in the rectosigmoid.\nFig. 2 Colonoscopy performed before surgery in 2014 showing extrinsic compression in the rectum by an endometriotic nodule.\nRev Bras Ginecol Obstet Vol. 40 No. 4/2018\nIntestinal Perforation due to Endometriosis during Pregnancy Carneiro et al. 237\n\n\nand that in many cases involving the rectovaginal septum or\nthe bowel, DIE can be appropriately managed without the\nsurgical excision of such lesions.\n8 A conservative approach\nseems justi fied in younger patients wishing to conceive,\nalthough segmental bowel resection and anastomosis result\nin better outcomes, without interfering in pregnancy rates.\n5\nThis may have been the case when our patient was first\noperated, as she was not infertile. Her clinical history,\nhowever, showed intestinal symptoms (dyschezia), and the\nmedical treatment she underwent apparently was not\nenough to stop the progression of her intestinal endometri-\nosis. In addition, the colonoscopy had already showed steno-\nsis probably related to extrinsic compression. Unfortunately,\nnone of the previous surgeries were performed by a multi-\ndisciplinary endometriosis team. It is advisable to refer\nwomen with DIE to a center of expertise in endometriosis\nthat can offer multidisciplinary management, as the surgery\nis associated with signi ficant complication rates.\n2\nDisastrously, intestinal rupture during pregnancy oc -\ncurred. Although rare, intestinal obstruction due to endo-\nmetriosis during pregnancy may happen, with rather severe\ncomplications. Unfortunately, DIE might be overlooked, as\nsymptoms can be elusive and unspeci fic, and intestinal\nlesions may be missed even during laparoscopy. Further-\nmore, smaller deep lesions, especially at the level of the\nsigmoid, may be missed during the diagnostic workup and\nlaparoscopy. Last, but not least, one expects endometriosis to\nregress during pregnancy.\n2,9 Regrettably, there may be more\ncases, as many may not have been published. We have\nreceived personal communication of two other cases of\nintestinal rupture after controlled ovarian stimulation for\nin vitro fertilization (IVF). As a matter of fact, acute compli-\ncations of endometriosis occurring during pregnancy remain\nrare. Setúbal et al\n9 published a review on intestinal compli-\ncations caused by DIE either during pregnancy or IVF. Their\nliterature search revealed 12 articles describing 12 compli-\ncations related to the progression of DIE during pregnancy,\nand 1 article reporting 6 cases of bowel occlusion during IVF.\nSurgery is not mandatory in all cases, and when infertility is\ninvolved, IVF appears to be the best option.\n2\nWomen presenting with intestinal symptoms and those\nwith previous endometriosis surgery are at increased risk of\nbowel resection. The decision to perform surgery in this\nsetting, however, remains mainly clinical. Available pub-\nlished data on the long-term outcomes reveals a cumulative\nprobability of pain recurrence between 20% and 40%, and\nneed of another surgical procedure between 15% and 20%.\nTherefore, the decision to perform radical or conservative\nsurgery or no surgical intervention at all should be evaluated\nindividually.\n10 Accurate information on the benefits and risks\ninvolved in the procedure versus treatment without bowel\nresection should be available to all patients. The manage-\nment of women with bowel endometriosis remains a chal-\nlenge due to the lack of published studies evaluating the real\nrisk of symptom recurrence, the effects on fertility, and the\nneed for further surgical interventions. Decisions, however,\nshould be individualized to the needs of each woman after\nproviding information on the potential bene fits, harms, and\ncosts of each treatment alternative, as well as after evaluating\nthe presence of pelvic pain, the woman ’s age, lesion location,\nand previous treatments.\n10 In such a setting, the evaluation\nby a multidisciplinary endometriosis team is a fundamental\nstep in order to achieve successful results.\nConflicts to Interest\nThe authors have no con flicts of interest to disclose.\nAcknowledgments\nThe authors wish to acknowledge that the surgeon respon-\nsible for the case herein reported was Dr. Ivone Dirk de\nSouza Filogonio. Unfortunately, she passed away in Decem-\nber 2015. For over 30 years, Dr. Filogonio was responsible for\nhundreds of surgeries, and in the past 20 years, she devoted\nmost of her time to the development of our multidisciplin-\nary endometriosis team.\nReferences\n1 Koninckx PR, Ussia A, Adamyan L, Wattiez A, Donnez J. Deep\nendometriosis: de finition, diagnosis, and treatment. Fertil Steril\n2012;98(03):564–571. Doi: 10.1016/j.fertnstert.2012.07.1061\n2 Dunselman GA, Vermeulen N, Becker C, et al; European Society of\nHuman Reproduction and Embryology. ESHRE guideline: man-\nagement of women with endometriosis. Hum Reprod 2014;29\n(03):400–412. Doi: 10.1093/humrep/det457\n3 De Cicco C, Corona R, Schonman R, Mailova K, Ussia A, Koninckx P .\nBowel resection for deep endometriosis: a systematic review. BJOG\n2011;118(03):285–291. Doi: 10.1111/j.1471-0528.2010.02744.x\n4 Meuleman C, Tomassetti C, D ’Hoore A, et al. Surgical treatment\nof deeply in filtrating endometriosis with colorectal involvement.\nHum Reprod Update 2011;17(03):311–326. Doi: 10.1093/humupd/\ndmq057\n5 Abrão MS, Petraglia F, Falcone T, Keckstein J, Osuga Y, Chapron C.\nDeep endometriosis infiltrating the recto-sigmoid: critical factors\nto consider before management. Hum Reprod Update 2015;21\n(03):329–339. Doi: 10.1093/humupd/dmv003\n6 Carneiro MM, Filogônio ID, Costa LM, de Ávila I, Ferreira MC.\nClinical prediction of deeply in filtrating endometriosis before\nsurgery: is it feasible? A review of the literature. BioMed Res\nInt 2013;2013:564153\n7 Milone M, Mollo A, Musella M, et al. Role of colonoscopy\nin the diagnostic work-up of bowel endometriosis. World J\nGastroenterol 2015;21(16):4997 –5001. Doi: 10.3748/wjg.v21.\ni16.4997\n8 Acién P, Núñez C, Quereda F, Velasco I, Valiente M, Vidal V. Is a\nbowel resection necessary for deep endometriosis with rectova-\nginal or colorectal involvement? Int J Womens Health 2013;\n5:449–455. Doi: 10.2147/IJWH.S46519\n9 Setúbal A, Sidiropoulou Z, Torgal M, Casal E, Lourenço C, Koninckx\nP. Bowel complications of deep endometriosis during pregnancy\nor in vitro fertilization. Fertil Steril 2014;101(02):442 –446. Doi:\n10.1016/j.fertnstert.2013.11.001\n10 Carneiro MM, Costa LMP, Ávila I. 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