{"paper_id":"03c73f67-264e-4048-98ee-135bba01b185","body_text":"Introduction\n!\nEndometriosis is a chronic disease that affects 4 –\n30 % of all women of child-bearing age. The preva-\nlence is even higher among women with infertili-\nty (up to 50 %) [1]. Dysmenorrhea is the main clin-\nical symptom of the disease, which is character-\nised by the presence of endometrial cells outside\nof the uterus. Endometriosis commonly occurs\nsuperficially in the peritoneal membrane howev-\ner it can also infiltrate deeply into e.g. the bladder,\nrectum and other bowel segments. Serious ma-\nternal pregnancy complications mostly occur in\nassociation with this deeply infiltrating form of\nthe disease.\nAfter data adjustment for age and gestational age\nwomen with endometriosis have tubal pregnan-\ncies almost three times more often (odds ratio\n2.7 [95 % confidence interval 1.09 –6.72]), and al-\nmost twice as many miscarriages (1.76 [1.44 –\n2.15]) compared to women without endometrio-\nsis [2]. The same study, with a study population of\n5375 patients, found increased risk of placenta\npraevia (OR 2.24), antenatal haemorrhage of un-\ncertain cause (OR 1.67) and postpartum haemor-\nrhage (OR 1.26) [2].\nEndometriosis does not always regress during\npregnancy. Endometrial decidualisation in partic-\nular is responsible for many of the complications\noccurring during pregnancy. Increased progester-\none production is causative [3]. The risk of endo-\nmetriosis complications is particularly high in the\nsecond half of pregnancy and during labour.\nRisks described in the literature include sponta-\nneous intraabdominal haemorrhage, uterine rup-\nture, bowel perforation, endometriosis cyst perfo-\nration and ovarian torsion in the presence of ovar-\nian endometrioma.\nAbstract\n!\nThe incidence of endometriosis is increasing. Par-\nticularly during pregnancy and labour, clinicians\nshould be alert to possible endometriosis-associ-\nated complications or complications of previous\nendometriosis treatment, despite a low relative\nrisk. In addition to an increased rate of early mis-\ncarriage, complications such as spontaneous bow-\nel perforation, rupture of ovarian cysts, uterine\nrupture and intraabdominal bleeding from decid-\nualised endometriosis lesions or previous surgery\nare described in the literature. Unfavourable neo-\nnatal outcomes have also been discussed. We re-\nport on an irreducible ovarian torsion in the 16th\nweek of pregnancy following extensive endome-\ntriosis surgery, and an intraabdominal haemor-\nrhage due to endometriosis of the bowel in the\n29th week of pregnancy.\nZusammenfassung\n!\nDurch die zunehmende Inzidenz der Endometri-\nose muss, trotz des geringen relativen Risikos, ge-\nrade während Schwangerschaft und Entbindung\nbei diesen Patientinnen an eine Komplikation\ndurch Endometriose oder vorherige Therapie ge-\ndacht werden. Neben einer erhöhten Abortrate in\nder Frühschwangerschaft werden in der Literatur\nKomplikationen wie spontane Darm- und Ovari-\nalzystenruptur, Uterusruptur sowie intraabdomi-\nnale Blutungen durch dezidualisierte Endome-\ntrioseherde oder vorangegangene Operationen\nbeschrieben. In der Literatur wird sogar ein un-\ngünstigeres neonatales Outcome diskutiert. Wir\nberichten über eine nicht mehr detorquierbare\nStieldrehung in der 16. SSW nach ausgedehnter\nEndometrioseoperation und eine intraabdomina-\nle Blutung bei Darmendometriose in der 29. SSW.\nEndometriosis-associated Maternal Pregnancy\nComplications – Case Report and Literature Review\nMaternale Endometriose-assoziierte Schwangerschaftskomplikationen –\nFallbericht und Literaturübersicht\nAuthors J. Petresin, J. Wolf, S. Emir, A. Müller, A. S. Boosz\nAffiliation Frauenklinik, Städtisches Klinikum, Karlsruhe\nKey words\nl\" endometriosis\nl\" pregnancy\nl\" complication\nl\" delivery\nl\" decidualization\nl\" intraabdominal bleeding\nSchlüsselwörter\nl\" Endometriose\nl\" Schwangerschaft\nl\" Komplikation\nl\" Hämoperitoneum\nreceived 21. 10. 2015\nrevised 12. 1. 2016\naccepted 17. 1. 2016\nBibliography\nDOI http://dx.doi.org/\n10.1055/s-0042-101026\nGeburtsh Frauenheilk 2016; 76:\n902–905 © Georg Thieme\nVerlag KG Stuttgart · New York ·\nISSN 0016‑5751\nCorrespondence\nDr. med.\nAlexander Stephan Boosz\nStädtisches Klinikum\nFrauenklinik\nMoltkestraße 90\n76133 Karlsruhe\nalexander.boosz@\nklinikum-karlsruhe.de\n902\nPetresin J et al. Endometriosis-associated Maternal Pregnancy … Geburtsh Frauenheilk 2016; 76: 902 –905\nDeutsche Version unter:\nhttp://dx.doi.org/\n10.1055/s-0042-101026\nGebFra Science\n\n\nCase 1\n!\nA 33-year-old patient (gravida III, para I) had previously had ex-\ntensive endometriosis surgery in 2009. Amongst other proce-\ndures she had had an extensive peritonectomy for peritoneal en-\ndometriosis. She subsequently underwent unsuccessful fertility\ntreatment. She ultimately fell pregnant naturally in 2011 and\nthe child was born in 2012 by caesarean section without compli-\ncation. Her subsequent pregnancy, the one in question, was also\nnatural.\nThe patient presented to us from an external hospital at 15 + 4\nweeks gestation with severe left-sided lower abdominal pain that\nhad been present for 3 days. Ultrasound showed an adnexal mass\nmeasuring approx. 60 × 40 mm. Symptoms improved with con-\nservative management, and an expectant approach with analge-\nsia was agreed on with the patient. After 2 days the patient ʼs\nsymptoms had not yet resolved and she was transferred to our\nhospital. On ultrasound the adnexal mass was found to have\ngrown to a diameter of approx. 80 mm, the structure appearing\nmostly haemorrhagic. Under suspicion of a progressively enlarg-\ning, haemorrhagic ovarian cyst with persistent symptoms the de-\ncision was made to perform a laparoscopy.\nIntraoperatively the uterus was appropriately enlarged for the\npregnancy and the right adnex was normal. The left adnex was\nobviously enlarged, with black discolouration, and was adherent\nto the pelvic wall and sigmoid colon. After adhesiolysis and mobi-\nlisation of the sigmoid a necrotic adnexal torsion was found\n(l\n\" Fig. 1 a and b). Detorsion was unsuccessful. The left broad liga-\nment had evidently been broken through, and the left adnex was\ntwisted 3 times on its long axis both in the region of the ovarian\nsuspensory ligament and the ovarian ligament ( l\n\" Fig. 1 a). Be-\ncause of the adnexal enlargement it was not possible to perform\ndetorsion through the defect in the broad ligament. A left adnex-\nectomy was thus performed ( l\n\" Fig. 2). The patient was dis-\ncharged home on the 4th postoperative day after an uneventful\npostoperative course. Fetal ultrasound before discharge was nor-\nmal, the patient delivered via repeat caesarean section in the\n39th week of gestation.\nCase 2\n!\nThis 25-year-old patient was admitted at 27 + 1 weeks gestation\nwith vaginal bleeding and preterm labour. On history she was\nnoted to have had a laparoscopic endometriosis operation in\n2011 and a previous appendicectomy. At laparoscopy in an exter-\nnal centre endometriosis was noted between the rectum and\nposterior uterine wall but left in situ since consent to treat it op-\neratively was lacking.\nInitially placental abruption was suspected, however this was not\nconfirmed and RDS prophylaxis with celestan as well as tocolysis\nwith fenoterol bolus was carried out. Ultrasound, laboratory pa-\nrameters and repeated CTGs showed no abnormalities. The pa-\ntientʼs symptoms improved under tocolysis and on the third day\nof admission had completely resolved, including the bleeding.\nCTG and cervical findings were persistently normal and she was\ndischarged.\nAt 28 + 2 weeks gestation she presented again, this time as an\nemergency, again with unusually heavy vaginal bleeding. The\ncervical length was 27 mm without funneling. She was admitted\nand tocolysis commenced with a partusisten bolus. On ultra-\nsound there was again no evidence of placental abruption. The\nfollowing day the patient was noted to have extremely severe ab-\ndominal pain that made micturition impossible. There was still\nno ultrasound evidence of placental abruption and both fetal\nheart rate and doppler indices were normal. Clinical signs were\nhowever suggestive of peritonitis, with extreme abdominal pain\nFig. 1 a and ba Case 1 – left adnexal torsion: The mass is twisted three\ntimes on its long axis between the ovarian suspensory ligament and the\novarian ligament. Detorsion was impossible due to the size of the mass. The\nsurgical instrument on the left is in the previously fenestrated broad liga-\nment. b Case 1 – haemorrhagic left adnex: Haemorrhagic infarction sec-\nondary to torsion. The surface is ruptured on attempted detorsion with\nblunt instruments.\nFig. 2 Case 1 – postoperative status, left pelvic wall: The left adnex has\nbeen removed. Excision was performed at the ovarian suspensory ligament\n(at bottom left of picture) and the ovarian ligament (top right).\n903\nPetresin J et al. Endometriosis-associated Maternal Pregnancy … Geburtsh Frauenheilk 2016; 76: 902 –905\nCase Report\n\n\nand muscular defence, circulatory instability and raised CRP. At\nthis point vaginal bleeding had stopped. The patient ʼs condition\nwas considered life-threatening and in the presence of recurrent\nvaginal bleeding of unknown cause the decision was made to\nperform a caesarean section despite the early gestation. Since\nthe aetiology of the acute abdomen was unknown it was decided\nto operate under general anaesthesia. At surgery an extensive\nhaemoperitoneum was found. After delivery of the child some\ndark blood clot was found in the uterus at the placental margins\nindicating a marginal sinus haemorrhage or early, partial\nplacental abruption. The child was delivered without complica-\ntion and after closure of the uterus the abdomen was explored\nto identify the source of the bleeding. Numerous actively bleed-\ning serosal defects were found on the posterior uterine wall and\nanterior surface of the rectum, and a torn adhesion was found be-\ntween bowel (sigmoid colon/rectum) and uterus. Attempted hae-\nmostasis using sutures was unsuccessful due to tissue vulnerabil-\nity. The posterior uterine wall was thus covered with haemostatic\nfibrin glue and compression applied. A Robinson drain was also\ninserted. There were no postoperative complications, blood\ntransfusion was not necessary and the patient was discharged\non the 5th postoperative day.\nDiscussion\n!\nCase 1\nTo our knowledge this is the first case report of an endometriosis\ncomplication in pregnancy occurring 5 years after previous sur-\ngery. It is conceivable that deperitonealisation ventral and dorsal\nof the left ovary had been performed. During surgery the ovary\nmay have been fixed temporarily to the abdominal wall to assist\nthe complicated operation. It is impossible to exactly estimate\nwhen the torsion occurred. The possibly long-standing torsion\nmay only have become symptomatic when the cyst increased in\nvolume in addition to uterine enlargement.\nSince ovary and Fallopian tube had completely twisted around\nthe axis between the uterine suspensory ligament and the ovar-\nian ligament adnexectomy was the only option. In view of the ex-\ntreme pain the patient was experiencing we thought laparoscopy\nwas indicated despite the minimally increased risk of intrauter-\nine fetal death [4].\nCase 2\nThis case illustrates a rare but typical endometriosis-associated\npregnancy complication. Hospital admission and RDS prophy-\nlaxis were undertaken in view of vaginal bleeding of uncertain\ncause. Within a few hours the pain had become extreme and\nthere were clinical signs of an acute abdomen, together clearly in-\ndicating the need for further investigation. Laparoscopy may also\nhave identified the problem, though the size of the uterus may\nhave been limiting [5]. In the context of recent onset vaginal\nbleeding in association with acute, severe pain we felt immediate\ndelivery of the baby was indicated since the patient ʼs condition\nwas potentially life-threatening and the causative pathology still\nunknown.\nIntraabdominal bleeding\nSpontaneous peritoneal bleeding occurs in the second half of\npregnancy, during labour and occasionally postpartum. The most\nimportant symptoms are acute or subacute abdominal pain fol-\nlowed by hypovolaemic shock and fetal distress [6]. The largest\nstudy on the incidence of haemoperitoneum in pregnancy is a\nretrospective analysis of 800 women from a period of 5 years.\nThe study describes three women (0.38 %) with significant intra-\nabdominal bleeding during the third trimester due to endome-\ntriosis [7]. There are also a few reports of stillbirths [8] and early\nneonatal deaths [9] due to fulminant haemoperitoneum causing\nhypovolaemic shock.\nIn pregnant women with the triad of a history of previous endo-\nmetriosis, severe abdominal pain and a fall in haemoglobin, in-\ntraabdominal haemorrhage must be considered [10]. Endome-\ntriosis can also rarely result in haemoperitoneum in non-gravid\npatients [11].\nEndometrioma\nThe incidence of adnexal tumours in pregnancy is approximately\n4 % [12], the figure varying between studies. 11.5 % of these tu-\nmours are endometriomas [13]. It is not possible to estimate the\nincidence of endometrioma-associated complications as there\nare too few cases reported. To date there have been four case re-\nports of perforated endometrioma during pregnancy [14]. Symp-\ntoms are those of intraabdominal bleeding.\nBowel perforation\nThe incidence of this complication also remains unknown. A re-\nview article on the topic found 12 cases reports of bowel perfora-\ntion during pregnancy that were caused by endometriosis [15].\nAll cases presented with an acute abdomen. All patients had\nemergency operations, either a Hartmann ʼs procedure or a seg-\nmental resection. Sites of perforation were as follows: 2 × small\nbowel, 1 × coecum, 3 × appendix and 6 × rectum and sigmoid co-\nlon [15]. Bowel perforation is thought to be the result of in-\ncreased traction on adherent endometriosis lesions by the grow-\ning uterus in combination with intraintestinal pressure from fae-\nces [16].\nUterine rupture\nSpontaneous uterine rupture during pregnancy is very rare in the\ncontext of adenomyosis without previous uterine surgery; two\ncase reports exist [17, 18]. Recent publications describe intrapar-\ntum uterine rupture following previous surgery for cystic adeno-\nmyosis [19, 20]. There is no clinical evidence supporting the re-\nsection of adenomyosis to improve fertility. The danger of uterine\nrupture must be considered in future patient care, especially\nwhen surgery has resulted in larger myometrial defects [21]. An\noptimal myometrial thickness of 9 –15 mm following resection is\ndescribed [22].\nSurgery during pregnancy\nSymptomatic ovarian cysts are operated most commonly at the\nend of the first and beginning of the second trimesters [4]. In a\nretrospective analysis one third of cysts became evident because\nof ovarian torsion [4]. Laparoscopic access and operative manage-\nment is possible up until the 26th to 28th week of gestation, de-\npendant on the surgeon ʼs experience [23].\nNo general recommendation can be made to treat endometriosis\noperatively during or before pregnancy in order to avoid this\ncomplication, since there is no evidence that this improves preg-\nnancy or pregnancy outcome [24].\n904\nPetresin J et al. Endometriosis-associated Maternal Pregnancy … Geburtsh Frauenheilk 2016; 76: 902 –905\nGebFra Science\n\n\nConclusion\n!\nThe complications described here are rare (approx. 0.4 %). A com-\nprehensive history including symptoms of endometriosis and\nprevious operations is decisive in the differential diagnosis. The\nrisk of these endometriosis-associated complications is deter-\nmined by endometriosis severity (superficial or deep infiltrating)\nas well as the extent of previous surgery. When the clinical pre-\nsentation is unclear further investigation must be undertaken\ndependant on gestation and including diagnostic/operative lapa-\nroscopy when indicated. Alternatively there should be a low\nthreshold for caesarean section, particularly at advanced gesta-\ntions. Elective caesarean section may be indicated for both com-\npletely operated endometriosis and incompletely operated, deep\ninfiltrating endometriosis. However, a general recommendation\ncan not be made.\nConflict of Interest\n!\nNone.\nReferences\n1 Burghaus S, Klingsiek P, Fasching PA et al. Risk factors for endometriosis\nin a german case-control study. Geburtsh Frauenheilk 2011; 71: 1073 –\n1079\n2 Kmietowicz Z. Endometriosis is linked to greater risk of complications\nin pregnancy and birth, study finds. BMJ 2015; 350: h3252\n3 Zaytsev P, Taxy JB. Pregnancy-associated ectopic decidua. Am J Surg\nPathol 1987; 11: 526 –530\n4 Koo YJ, Lee JE, Lim KT et al. A 10-year experience of laparoscopic sur-\ngery for adnexal masses during pregnancy. Int J Gynaecol Obstet\n2011; 113: 36 –39\n5 Juhasz-Boss I, Solomayer E, Strik M et al. Abdominal surgery in preg-\nnancy–an interdisciplinary challenge. Dtsch Arztebl Int 2014; 111:\n465–472\n6 Brosens IA, Fusi L, Brosens JJ. Endometriosis is a risk factor for sponta-\nneous hemoperitoneum during pregnancy. Fertil Steril 2009; 92:\n1243–1245\n7 Katorza E, Soriano D, Stockheim D et al. 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Cancro 1967; 20: 400\n–410\n18 Kawabara H, Yazaki H, Hosobe S. [Endometriosis of the uterine wall and\nspontaneous rupture of the pregnant uterus]. Jpn J Med Sci Biol 1962;\n48: 177–180\n19 Fettback PB, Pereira RM, Domingues TS et al. Uterine rupture before the\nonset of labor following extensive resection of deeply infiltrating en-\ndometriosis with myometrial invasion. Int J Gynaecol Obstet 2015;\n129: 268–270\n20 Dim CC, Agu PU, Dim NR et al. Adenomyosis and uterine rupture during\nlabour in a primigravida: an unusual obstetric emergency in Nigeria.\nTrop Doct 2009; 39: 250 –251\n21 Pepas L, Deguara C, Davis C. Update on the surgical management of ad-\nenomyosis. Curr Opin Obstet Gynecol 2012; 24: 259 –264\n22 Otsubo Y, Nishida M, Arai Y et al. Association of uterine wall thickness\nwith pregnancy outcome following uterine-sparing surgery for diffuse\nuterine adenomyosis. Aust N Z J Obstet Gynaecol 2015; DOI: 10.1111/\najo.12419\n23 Pearl J, Price R, Richardson W et al. Guidelines for diagnosis, treatment,\nand use of laparoscopy for surgical problems during pregnancy. Surg\nEndosc 2011; 25: 3479 –3492\n24 Leone Roberti Maggiore U, Ferrero S, Mangili G et al. A systematic re-\nview on endometriosis during pregnancy: diagnosis, misdiagnosis,\ncomplications and outcomes. Hum Reprod Update 2016; 22: 70 –103\n905\nPetresin J et al. Endometriosis-associated Maternal Pregnancy … Geburtsh Frauenheilk 2016; 76: 902 –905\nCase Report","source_license":"public-domain-us","license_restricted":false}