{"paper_id":"8564ffd5-c048-4e67-bde8-52a2feff3a25","body_text":"C A S E R E P O R T Open Access\nUterine pseudoaneurysm on the basis of\ndeep infiltrating endometriosis during\npregnancy-a case report\nTibor Andrea Zwimpfer 1,2* , Cécile Monod 1, Katharina Redling 1, Heike Willi 1, Martin Takes 3,\nBernhard Fellmann-Fischer 1, Gwendolin Manegold-Brauer 1 and Irene Hösli 1\nAbstract\nBackground: Pseudoaneurysm of the uterine artery (UPA) is a rare cause of potentially life-threatening hemorrhage\nduring pregnancy and puerperium. It is an uncommon condition that mainly occurs after traumatic injury to a\nvessel following pelvic surgical intervention, but also has been reported based on underlying endometriosis. There\nis an increased risk of developing UPA during pregnancy. Diagnosis includes clinical symptoms, with severe\nabdominal pain and is confirmed by sonographic or magnetic resonance imaging (MRI). Due to its potential risk of\nrupture, with a subsequent hypovolemic maternal shock and high fetal mortality, an interdisciplinary treatment\nshould be considered expeditiously.\nCase presentation: We present the case of a 34-year old pregnant symptomatic patient, where a large UPA was\ndetected at 26 weeks, based on deep infiltrating endometriosis (DIE). The UPA was successfully treated by selective\narterial embolization. After embolization, the pain decreased but the woman still required intravenous analgesics\nduring follow-up. At 37 weeks she developed a sepsis from the intravenous catheter which led to a cesarean\nsection and delivery of a healthy boy. She was discharged 10 days postpartum.\nConclusions: UPA should be considered in pregnant women with severe abdominal and pelvic pain, once other\nobstetrical factors have been excluded. DIE might be the underlying diagnosis. It is a rare but potentially life-\nthreatening condition for mother and fetus.\nKeywords: Uterine pseudoaneurysm, Deep infiltrating endometriosis, Pregnancy\nBackground\nUterine pseudoaneurysm (UPA) is a condition in which\nthe arterial vessel wall has lost intraluminal continuity and\nblood accumulates between the two outer layers of the\nartery. It can present with severe abdominal and pelvic\npain, and sonographic imaging or magnet resonance\nimaging (MRI) can detect a pulsatile growing mass. UPA\noccurs mainly after a traumatic injury of the vessel follo-\nwing pelvic surgical intervention, but rarely, it is based on\nsevere endometriosis as DIE (Deep infiltrating endome-\ntriosis) [1–5]. The main causes are gynecological interven-\ntions, such a myomectomy, treatment of endometriosis,\novarian puncture or cystectomies, and obstetrical inter-\nventions, such as cesarean section, curettage, and vacuum\nor forceps extraction [ 3–9]. Furthermore, there is an\nincreased risk of developing UPA during pregnancy.\nDue to the potential risk of rupture, with subsequent\nhypovolemic shock of the mother and a high fetal mor-\ntality, the diagnosis of UPA in pregnancy requires urgent\n© The Author(s). 2021 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License,\nwhich permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give\nappropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if\nchanges were made. The images or other third party material in this article are included in the article's Creative Commons\nlicence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons\nlicence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain\npermission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.\nThe Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the\ndata made available in this article, unless otherwise stated in a credit line to the data.\n* Correspondence: tibor.zwimpfer@unibas.ch\n1Department of Obstetrics and Gynecology, University Hospital of Basel,\nBasel, Switzerland\n2Department of Biomedicine, University Hospital of Basel and University\nBasel, Basel, Switzerland\nFull list of author information is available at the end of the article\nZwimpfer et al. BMC Pregnancy and Childbirth          (2021) 21:282 \nhttps://doi.org/10.1186/s12884-021-03753-1\n\ninterdisciplinary treatment. The standard of care is a se-\nlective arterial embolization of the uterine artery by the\ninterventional radiologist, which has a good risk-benefit\nprofile [ 10, 11].\nHere, we present the observation of a pregnant patient\nwith a successfully treated symptomatic UPA that oc-\ncurred in the second trimester, on the basis of a DIE in\nthe left uterine artery and cervix.\nCase presentation\nThe 34-year old, first gravida, was admitted to our ob-\nstetrical department by ambulance at 23 + 0 weeks of\ngestation (WG) with progressive severe pain over 24 h in\nthe left lower abdomen irradiating to the rectum and the\nvagina. The previous day, she had an unremarkable clin-\nical and sonographic examination and a normal labora-\ntory investigation. Her past medical history included a\nconization due to cervical dysplasia, dysmenorrhea and\ndyspareunia with suspected endometriosis, and the use\nof a combined oral contraceptive for 16 years prior to\nthe current pregnancy. The pregnancy occurred\nspontaneously, with a single fetus. The otherwise healthy\npatient showed pain on palpation in the two lower ab-\ndominal quadrants: speculum examination revealed cer-\nvical ectopy and two black dots were visible at six\no’clock. The vital signs were normal with an unremark-\nable pulse, respiratory rate, body temperature, blood\npressure, and the hemoglobin was stable at 129 g/l. The\nlaboratory tests and urine analysis showed no signs of\ninfection. The cardiotocogram (CTG) was normal with-\nout contractions. Cervical length was 34 mm, measured\nby transvaginal ultrasound. Fetal sonography and Dop-\npler studies revealed normal biometry with a fetus ap-\npropriate for gestational age at 42nd percentile, normal\namniotic fluid, a posterior wall placenta without signs of\nhematoma and a normal uteroplacental resistance. Cau-\ndal and adjacent to the left ovary, a solid, ill-defined ad-\nnexal mass of 40x45mm and moderate blood flow was\ndetected (Fig. 1 a, b). On MRI, the adnexal mass was\nseen and was suggestive of endometriosis (Fig. 2 a and\nb). There were no signs of intraabdominal free fluid or\nkidney stones.\nFig. 1 Development of the UPA over 2 weeks by ultrasound. Visit 1 with a. Ultrasound image of the ill-defined solid mass in the adnex at the\ninitial presentation at 23 + 2 gestational weeks. The arrows mark the outer margins of the lesion. An endometrioma is not clearly visible. b.\nCorresponding Doppler image of the left adnexa showing moderate blood flow (Color Score 3) in the lesion. Visit 2 with cUltrasound Image of\nthe same lesion on the follow up at 25 + 2 gestational weeks. On the lower right there is an unilocular mass suggestive of endometrioma. In the\ncenter there is a pulsating vessel (UPA) of about 2 cm with most likely haematoma surrounding the UPA and d. corresponding Doppler image\nconfirming blood flow in the vessel.\nZwimpfer et al. BMC Pregnancy and Childbirth          (2021) 21:282 Page 2 of 7\n\nThe patient was hospitalized for monitoring and anal-\ngesic therapy. The pelvic pain persisted despite intraven-\nous (IV) opiate therapy. Additionally, the patient\ncomplained of newly occurring dyschezia and a single\nepisode of brown vaginal discharge. A follow-up MRI\nafter 6 days, at 24 + 1 WG showed progression of the le-\nsion, with expansion into the rectovaginal space (Fig. 2 c\nand d). The patient remained hospitalized for maternal\nand fetal monitoring and received continuous analgesia\nand steroids for fetal lung maturation induction with\nFig. 2 Timeline of the development of the endometriosis nodule and pseudoaneurysm of the uterine artery (UPA), from transvaginal ultrasound\n(TVUS) and magnetic resonance imaging (MRI). a: T1 weighted transverse MRI showing a lesion (2 cm × 2 cm) caudal adjacent to the left ovary,\nsuspected to be an endometriosis nodule; b: Dynamic MR angiography (TWIST) showing a small process of the left uterine artery.; c.T1 weighted\ntransverse MRI showing a progressing lesion (3.8 cm × 2 cm) with expansion to the rectovaginal space with d. a corresponding growing alteration\nof the left uterine artery in the TWIST.; e. T1 weighted transverse MRI with an identifiable UPA (2.5 cm × 1.5 cm) and the confirmation in the f.\nTWIST.; g. TVUS showing the UPA (left) (black arrow) and the endometriosis nodule (white arrow) one day after embolization; h: TVUS follow-up\nultrasound a week after embolization, showing the UPA left (black arrow) and endometriois nodule (white arrow)\nZwimpfer et al. BMC Pregnancy and Childbirth          (2021) 21:282 Page 3 of 7\n\ntocolysis as a precautionary measure. Sixteen days after\nhospitalization (25 + 3 WG), a pseudoaneurysm of the\nleft uterine artery (2.5 cm × 1.5 cm) was identified by\nultrasound (Fig. 1 c and d) and confirmed by MRI. (Fig.\n2 e and f). An interdisciplinary team of radiologist, feto-\nmaternal specialists, gynecologists and neonatologists\nconcluded that, based on the early gestational age, the\nprogression of the lesion and in view to the persistent\npain, a selective embolization of the left uterine artery\nwould be the preferred management. The neonatologist\nteam was prepared for an emergency cesarean section in\ncase of any complications. The intervention was carried\nout under local anesthesia with a retrograde access over\nthe left common femoral artery. No digital subtraction\nangiography was performed in order to minimize radi-\nation exposition. Embolization was achieved by selective\nocclusion of the aneurysm-feeding branch of the left\nuterine artery over a microcatheter by injection of 0.2 ml\nliquid embolic material (histoacryl/lipiodol 1:3). The es-\ntimated radiation exposure for the uterus was 0.4 mSv.\nSubsequently the contrast media had suspended in the\nUPA as indication for a sufficient occlusion. Further se-\nquential sonographic examinations confirmed normal\nfetal growth and Doppler flow. The embolized UPA\nshowed no vascularization and decreased to 2 cm × 1 cm\non sonography before hospital discharge (Fig. 2 g and h).\nThe pelvic pain improved but did not resolve completely\nand the patient received a peripherally inserted central\nvenous catheter (PICC) line and, temporarily, a patient-\ncontrolled analgesia pump. After 30 days (28 + 2 WG),\nshe was discharged to outpatient care with oral mor-\nphine in reserve and the PICC line in situ. A primary\ncesarean at 38 weeks was planned because of the risk of\nthe UPA rupturing during contractions. However, the\npatient presented herself at 37 weeks, with sepsis of un-\nknown etiology, a fever of 39.0 °C, tachypnea,\nhypotension, maternal tachycardia, a C-reactive protein\nof 23.8 mg/l and fetal tachycardia (200 beats per minute).\nAn emergency cesarean delivery was done under general\nanesthesia with antibiotic therapy (amoxicillin and clavu-\nlanic acid) and a healthy boy was born (2670 g) with\nApgar 6/7/8 and pH 7.31. Postoperatively, the patient\nneeded intensive care (IC) for 3 days. Blood cultures\nwere positive for Serratia macescens and Streptococcus\nanginosus, and according to the resistance tests, treat-\nment was changed to carbapenem IV. The patient was\ndischarged, together with the newborn, 10 days after the\ncesarean section. The infection was most likely caused\nby an infected PICC line, even though the results of the\nsmear tests and cultures were unremarkable.\nThe patient presented for follow-up at 6 weeks post-\npartum. She had lactation amenorrhea, persisting dys-\nchezia and newly developed hematochezia. Rectovaginal\nsonography and palpation identified an unchanged\nendometriosis node. We started suppressive therapy\nwith Desogestrel and scheduled a colonoscopy to ex-\nclude another origin for the hematochezia and an MRI\nfor staging. The MRI showed an endometriosis node\n3x2cm adjacent to the septum rectovaginale with expan-\nsion to the left ovary and in close proximity to the sig-\nmoid without infiltration according to an Enzian score\nA2, B1, C1. (Fig. 3 a and b) The restructuring operation\nof the symptomatic DIE is planned for 4 months after\nher delivery.\nDiscussion\nThis case illustrates the difficulty to diagnose the rare\nentity of a UPA during pregnancy. In our case, the ap-\npearance of the lesion changed over time and finally led\nto the correct diagnosis with the combination of sonog-\nraphy and MRI. There is an increased risk of developing\nor diagnosing UPA during pregnancy. It is assumed that\nthe physiological changes of the hormonal milieu and\ncardiovascular system, together with the pressure on the\nvessels promote the development of UPA [ 12]. Addition-\nally, the improvement of imaging technology and the\nfrequent ultrasounds during pregnancy increase the\nprobability of diagnosing a UPA.\nEndometriosis further increases the risk of UPA dur-\ning and after pregnancy, in particular DIE [ 13–15]. The\nendometrial implants demonstrate a non-location re-\nsponse to hormonal stimulation. Estrogens are a prolif-\nerating factor, and the hormone withdrawal results in\nabortive bleeding which is associated with pain. Add-\nitionally, inflammatory cell production is stimulated,\nresulting in pain and adhesions. Gestagen inhibits the in-\nflammatory reaction. In menopause, the decline of\novarian stimulation turns active endometriosis lesions\ninactive. Since pregnancy has a similar effect, with a\ndecline of ovarian stimulation and increasing gestagen\nlevels, a common assumption is that pregnancy tem-\nporarily cures endometriosis [ 16–18]. Recently, con-\nflicting data demonstrates preexisting endometriosis\ncauses pregnancy complications due to adhesions,\nchronic inflammation and intrusion of decidualized\nendometriosis [ 19–21]. Chronic inflammation makes\nthe vessels more vulnerable to lacerations [ 22]a n d\nadhesion can increase the stress on uterine-ovarian\nvessels [ 23]. The intrusion of decidualized endometri-\nosis can result in a perforation of the uterine-ovarian\nvessels and, because of persistent progesterone levels,\ndecidualization occurs with differentiation of mesen-\nchymal cells [ 24, 25]. A decrease in progesterone at\nthe end of the third trimester of pregnancy corre-\nlates with an increased expression of inflammatory\ncells, proteolytic degrad ation of the extracellular\nmatrix, cell death, and, finally, bleeding of the peri-\ntoneum [ 26, 27].\nZwimpfer et al. BMC Pregnancy and Childbirth          (2021) 21:282 Page 4 of 7\n\nIn this patient, the combination of preexisting DIE and\npregnancy probably caused the UPA. The close follow-\nup, with ultrasound and MRI, enabled us to detect the\ndevelopment of the UPA from the endometriosis node\nduring the second trimester. Previously, ruptured or\nunruptured UPA have been detected in pregnant pa-\ntients with a known history of endometriosis or previous\nsurgery [ 13–15]. Van Coppenollea et al. summarized six\ncases of UPA, based on previous appendectomy,\ncesarean section or surgically treated endometriosis [ 9].\nFeld et al. described even a case of a hemoperitoneum,\ncaused by a ruptured UPA based on endometriosis, but\nthe UPA was only detected postpartum [ 13]. Our patient\nwas symptom-free apart from occasional dyspareunia\nand dysmenorrhea because of a suspected rectovaginal\nendometriosis before pregnancy, as she was on hormo-\nnal contraception for 16 years until 6 months before\npregnancy. We presume that the pregnancy stimulated\nthe decidualization of the endometriosis, in particular of\nthe deep infiltrating rectovaginal node adjacent to the\nUPA. The chronic inflammation, in combination with\nthe decidualization, might have increased the stress on\nthe uterine artery resulting in the UPA. Fortunately, the\nUPA was diagnosed early, and the expedited treatment\npreserved the pregnancy and avoided preterm delivery.\nTranscatheter arterial embolization has been estab-\nlished as an effective technique for the management and\nprevention of obstetric and gynecologic hemorrhage [ 10,\n11, 15, 28–30]. Complications of transcatheter arterial\nembolization are extremely uncommon when it is per-\nformed by expert interventional radiologists. Its advan-\ntages include prevention of surgical risks, high success\nrates, low complication rates, and no significant impact\non future pregnancies and fertility [ 10, 11, 15, 28–30].\nThree reported cases in literature [ 11, 14, 30] and our\ncase suggest that successful unilateral uterine artery\nembolization is well tolerated by the fetus and therefore\nappears to be a safe and effective method to treat pseu-\ndoaneurysm during pregnancy without compromising\nuteroplacental perfusion. Moreover, in our case the esti-\nmated radiation exposure for the uterus was only 0.4\nmSv, which is far below any critical exposure rate for the\nfetus.\nOur patient suffered long-term from severe immobiliz-\ning pain, which was difficult to control. Furthermore,\nthe hospitalization and treatment were physically and\nemotionally very stressful. This raises the question re-\ngarding early diagnosis and treatment of such cases\nthrough monitoring pregnancies of patients with endo-\nmetriosis. Currently, there is no evidence that endomet-\nriosis has a significant effect on pregnancy outcome [ 31,\n32].; however, rare cases such as our case might be en-\ncountered in pregnancy and a data base of deep infiltrat-\ning endometriosis like that available at the Kepler\nUniversity Clinic together with the Foundation Endo-\nmetriosis Research (SEF), and with support of the\nDeutsche Gesellschaft für Gynäkologie und Geburtshilfe\n(DGGG) will be helpful in identifying patients at risk for\nsuch a complication and provide diagnostic and treat-\nment guidelines.\nConclusions\nUPA should be considered in pregnant women with se-\nvere abdominal and pelvic pain, once other obstetrical\nfactors have been excluded. Endometriosis can cause\nUPA during and after pregnancy, in particular DIE. It is\na rare but potentially life-threatening condition for the\nmother and fetus, as a rupture of the UPA will result in\nhemoperitoneum and hypovolemic, hemorrhagic shock.\nThe standard of care in a stable situation is selective ar-\nterial embolization, which has a good risk-benefit profile.\nThere is currently no evidence that endometriosis has a\nFig. 3 Abdominal magnetic resonance imaging (MRI) two months after the delivery a. T2 weighted transverse MRI and b. T2 weighted coronal\nMRI showed an endometriosis node 3x2cm (white arrow) adjacent to the septum rectovaginale with expansion to the left ovary and in close\nproximity to the sigmoid without infiltration according to an Enzian score A2, B1, C1\nZwimpfer et al. BMC Pregnancy and Childbirth          (2021) 21:282 Page 5 of 7\n\nharmful effect on the pregnancy outcome, therefore, no\nspecial monitoring of conventional pregnancies for pa-\ntients with endometriosis is required. Nevertheless,\nawareness should be raised among physicians, and simi-\nlar cases should be reported to establish treatment\nguidelines.\nAbbreviations\nCTG: Cardiotocogram; DIE: Deep infiltrating endometriosis; IC: Intensive care;\nIV: Intravenous; MRI: Magnetic resonance imaging; PICC: Peripherally inserted\ncentral venous catheter; TVUS: Transvaginal ultrasound; UPA: Uterine\npseudoaneurysm; WG: Weeks of gestation\nAcknowledgements\nNot applicable.\nAuthors’ contributions\n(I) Conception and design: TZ, KR, HW, GM. (II) Administrative support: IH, BF,\nGM, MT. (III) Collection and assembly of data: TZ, CM, MT. (IV) Data analysis\nand interpretation: TZ, CM, KR, HW. (V) Manuscript writing: all authors. (VI)\nFinal approval of manuscript: all authors.\nFunding\nNot applicable.\nAvailability of data and materials\nThe data and materials analyzed during the current case report are\npresented within the manuscript and available from the corresponding\nauthor.\nDeclarations\nEthics approval and consent to participate\nAccording to the ethics committee of Northwest and Central Switzerland\n(EKNZ) the project does not fall under the remit of the cantonal or federal\nlaw Human Research Act (HRA) because the project is not defined as a\nresearch project as per HRA Art. 2; therefore, an Institutional Review Board\napproval is not needed. The patient gave her written consent to participate\nin this case report and waived any claims. The anonymization of personal\ndata was guaranteed.\nConsent for publication\nWritten informed consent was obtained from the patient for publication of\nthis case report and any accompanying images. A copy of the written\nconsent is available for review by the Editor of this journal.\nCompeting interests\nThe authors Tibor Andrea Zwimpfer, Cécile Monod, Katharina Redling, Heike\nWilli, Martin Takes, Bernhard Fellmann-Fischer, Gwendolin Manegold-Brauer\nand Irene Hösli have no competing interest to disclose.\nAuthor details\n1Department of Obstetrics and Gynecology, University Hospital of Basel,\nBasel, Switzerland. 2Department of Biomedicine, University Hospital of Basel\nand University Basel, Basel, Switzerland. 3Department of Radiology and\nNuclear Medicine, University Hospital of Basel, Basel, Switzerland.\nReceived: 15 December 2020 Accepted: 24 March 2021\nReferences\n1. Ferrero S, Bogliolo S, Rossi UG, Baldi C, Valenzano Menada M, Ragni N, et al.\nUnusual complication of excision of pelvic endometriosis: pseudoaneurysm\nof the left uterine artery. Fertil Steril. 2010;93(1):264 –6. https://doi.org/10.101\n6/j.fertnstert.2009.02.002.\n2. 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Fertil Steril https://doi.org/ https://doi.org/10.1016/j.fertnstert.2017.\n07.019, 2017.\nPublisher’sN o t e\nSpringer Nature remains neutral with regard to jurisdictional claims in\npublished maps and institutional affiliations.\nZwimpfer et al. BMC Pregnancy and Childbirth          (2021) 21:282 Page 7 of 7","source_license":"CC0","license_restricted":false}