Pseudoaneurysm of the uterine artery is a rare complication of cesarean section. Delayed diagnosis and management may result in rapid and catastrophic postpartum hemorrhage and may necessitate hysterectomy or can even be fatal. A 28-year-old woman (gravida I, para I) presented with delayed postpartum hemorrhage 10 days after emergency cesarean section. Using transabdominal and transvaginal Color Doppler sonography, we detected a pseudoaneurysm in the posterior uterine wall, which was successfully treated with selective embolization distal and proximal to the lesion with platinum coils. In patients with delayed postpartum hemorrhage-especially after cesarean section-the rare possibility of uterine artery pseudoaneurysm must be kept in mind

Althoff, Christian E.

Henrich, Wolfgang

Hinkson, Larry

Pohlan, Julian

Wickmann, Ulrike

English

Institutional Repository of the Freie Universität Berlin

C A S E R E P O R TPseudo aneurysm of the uterine artery with arteriovenousfistula after cesarean section: A rare but sinister causeof delayed postpartum hemorrhageJulian Pohlan MD1 | Larry Hinkson MBBS, MRCOG, FRCOG, MD2 |Ulrike Wickmann MD2 | Wolfgang Henrich MD, PhD2 | Christian E. Althoff EBIR, MD11Department of Radiology, Campus CharitéMitte, Berlin, Germany2Department of Obstetrics, CharitéUniversitätsmedizin Berlin, Campus CharitéMitte, Berlin, GermanyCorrespondenceJulian Pohlan, Department of Radiology,Campus Charité Mitte, Luisenstraße 7, 10117Berlin, Germany.Email: julian.pohlan@charite.deLarry Hinkson, Department of Obstetrics,Charité Universitätsmedizin Berlin, CampusCharité Mitte, Berlin, Germany.Email: larry.hinkson@charite.deAbstractPseudoaneurysm of the uterine artery is a rare complication of cesarean section.Delayed diagnosis and management may result in rapid and catastrophic postpartumhemorrhage and may necessitate hysterectomy or can even be fatal. A 28-year-oldwoman (gravida I, para I) presented with delayed postpartum hemorrhage 10 daysafter emergency cesarean section. Using transabdominal and transvaginal ColorDoppler sonography, we detected a pseudoaneurysm in the posterior uterine wall,which was successfully treated with selective embolization distal and proximal to thelesion with platinum coils. In patients with delayed postpartum hemorrhage—especially after cesarean section—the rare possibility of uterine artery pseudo-aneurysm must be kept in mind.K E YWORD Scesarean section complication, Doppler ultrasound, embolization, postpartum hemorrhage,uterine artery pseudoaneurysm1 | CASE REPORTA 28-year-old patient (gravida I, para I) presented with heavy delayedpostpartum hemorrhage and lower abdominal pain, 10 days afteremergency cesarean section for failure to progress and suspectedchorioamnionitis. At the time of cesarean section, a 1200-mL bloodloss due to uterine atony was successfully managed with 800 μg ofmisoprostol rectally, IV (intravenous) infusion of 20 IU (internationalunits) of oxytocin, and 1 g of IV tranexamic acid. She was dischargedwith a hemoglobin level of 8.3 g/dL, which further dropped to 7.4 g/dLon readmission 10 days after the cesarean section.Portable bedside transabdominal and transvaginal ultrasoundexaminations with color Doppler (Philips Lumify with a C5-2 convexprobe and GE Voluson 730 with 5-13 MHz endocavitary probe) rev-ealed a sharply demarcated, hypoechoic 4-cm lesion at the posterioruterine wall entirely perfused on color Doppler US, consistent with apseudo aneurysm (Figure 1A,B). Contrast-enhanced computed tomog-raphy (CT) confirmed a 4-cm pseudoaneurysm at a segmental branchof the left uterine artery (Figure 1C). An emergency angiography andembolization was decided with the obstetric team ready for an emer-gency hysterectomy in case of uncontrollable rupture during the inter-ventional embolization procedure. Medical management at this stageincluded transfusion of one unit of red blood cells and 1 g of IV tran-examic acid. Antibiotic prophylaxis was given with 1.5 g cefuroximeintravenously.The intervention was performed under general anesthesia. Theright common femoral artery was accessed, and a crossover maneuverwas used to enter the left internal iliac artery for selective catheteriza-tion of the left uterine artery. Angiography provided additional infor-mation about the lesion, confirming a 4-cm pseudoaneurysm and anReceived: 10 April 2020 Revised: 2 June 2020 Accepted: 16 June 2020DOI: 10.1002/jcu.22890This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium,provided the original work is properly cited.© 2020 The Authors. Journal of Clinical Ultrasound published by Wiley Periodicals LLC.J Clin Ultrasound. 2021;49:265–268. wileyonlinelibrary.com/journal/jcu 265arteriovenous fistula component with rapid flow to a uterine vein(Figure 2A).The pseudoaneurysm was coiled distally and proximally and onthe side of the arteriovenous fistula using a total of 11 platinum coils.The intervention was technically successful, resulting in completeobstruction of blood flow into and from the pseudoaneurysm(Figure 2B). Transabdominal ultrasound examination (with a GE S10scanner and a convex 2-5 MHz probe) immediately after the emboli-zation confirmed complete collapse of the pseudoaneurysm. During afollow-up visit 4 weeks after embolization, the patient was asymp-tomatic, and transabdominal ultrasound revealed no reperfusion(Figure 2C). Delaying the next pregnancy for at least 12 months wasrecommended.2 | DISCUSSIONDelayed postpartum hemorrhage more than 24 hours after deliverycan have many underlying causes, and the differential diagnosis ischallenging. Common causes include uterine atony, retained productsof conception, vaginal tract trauma, and coagulation defects. Timelyrecognition of the rare but life-threatening uterine artery pseudo-aneurysm is critical for patient management due to its high risk ofrupture. Although most patients present early after cesarean section,a high index of suspicion is warranted as cases of delayed manifesta-tion have been reported.1Uterine artery pseudoaneurysm can develop secondary to traumaticinjury to the artery at the time of cesarean section. The wall of a pseudo-aneurysm is particularly fragile as the three layers of a true aneurysm(tunica, media, and adventitia) are absent, and rupture can lead to cata-strophic bleeding. Importantly, if delayed postpartum hemorrhage isoverlooked and instead misdiagnosed as retained products of conceptionand a therapeutic curettage is performed, this can even worsen thebleeding.1,2 Transabdominal and transvaginal ultrasound with colorDoppler are diagnostic. Examiners should look for a “To-and-fro” wave-form on duplex Doppler ultrasound.3 The “Yin-yang” sign can also bediagnostic on color Doppler examination .4 Angiography is both confir-matory and therapeutic. Most uterine artery pseudoaneurysms are suc-cessfully treated with gel foam and/or metallic coils.2 A case report inthe literature describes intra-arterial thrombin administration.5 Erikssonet al followed up patients after interventional treatment of uterine arterypseudoaneurysm and observed no case of infertility after embolization.6Embolization may also be performed bilaterally in women with massivebleeding.7 In resource-limited settings, surgical uterine artery ligation isan alternative to preserve fertility.8 Hysterectomy may have to be con-sidered in some patients with life-threatening hemorrhage.F IGURE 1 Imaging of the pseudoaneurysm. Transabdominal, A, and transvaginal, B, color Doppler sonograms showing the pseudoaneurysmwith the yin-yang sign. C, Sagittal reconstructed contrast-enhanced CT scan shows the contrast-filled pseudoaneurysm of the left uterine artery266 POHLAN ET AL.This case report aims at raising awareness of uterine artery pseu-doaneurysm as a potentially fatal underlying cause of postpartumhemorrhage. Endovascular treatment of uterine artery pseudo-aneurysms is effective and should be preferred to surgery.In patients with delayed postpartum hemorrhage—especially aftercesarean section—a uterine artery pseudoaneurysm should be suspectedand can be confirmed or ruled out using transabdominal and transvaginalcolor Doppler ultrasound. Contrast-enhanced CT or MRI may also behelpful in confirming the diagnosis and designing the treatment plan.Embolization of uterine pseudoaneurysm is technically feasibleand may prevent emergency hysterectomy in these patients. How-ever, data on the best choice of embolization material are still limited,and further research should focus on follow-up recommendations tomonitor reperfusion and plan future pregnancies.3 | CONCLUSIONBased on published evidence and the case presented here, we recom-mend color-coded Doppler transabdominal and transvaginal ultra-sound examination of the uterus with color Doppler is required inwomen presenting with delayed postpartum hemorrhage to rule oututerine artery pseudoaneurysm. Once diagnosed, urgent angiographicmanagement with an obstetric team on stand-by is warranted.ACKNOWLEDGMENTSThe authors thank BH for correcting and improving the manuscript.No funding was used for this case report.CONFLICT OF INTERESTThe authors declare that they have no competing interests.AUTHOR CONTRIBUTIONSChristian E. Althoff performed angiography. Larry Hinkson performedultrasound and co-wrote the manuscript. Wolfgang Henrich providedvaluable input for the manuscript. Julian Pohlan prepared manuscript andimages and corrected the manuscript. Ulrike Wickmann performed ultra-sound. All authors read and approved the final manuscript.DATA AVAILABILITYThe datasets used during the current case report are available fromthe corresponding author on reasonable request.F IGURE 2 A, Angiographic visualization of uterine pseudo aneurysm with arteriovenous fistula. B, Postembolization angiogram shows theplatinum coils. C, Transabdominal sonogram 4 weeks after embolization with no detectable color Doppler signalPOHLAN ET AL. 267ORCIDJulian Pohlan https://orcid.org/0000-0002-5189-4775REFERENCES1. Johannesson L, Mason J, Oehler MK. Uterine artery pseudoaneurysmbleeding one decade after caesarean section: a case report. J ObstetGynaecol Res. 2017;43(5):939-942.2. Moatti Z, Nisner T, Saini A, et al. Delayed postpartum haemorrhagesecondary to a ruptured uterine artery pseudo-aneurysm, successfullytreated by transarterial embolisation. Case Rep. 2011;2011:bcr0520114270. https://pubmed.ncbi.nlm.nih.gov/22674937/.3. Mahmoud MZ, Al-Saadi M, Abuderman A, et al. "to-and-fro" waveformin the diagnosis of arterial pseudoaneurysms. World J Radiol. 2015;7(5):89-99.4. Corvino A, Catalano O, de Magistris G, et al. Usefulness of dopplertechniques in the diagnosis of peripheral iatrogenic pseudoaneurysmssecondary to minimally invasive interventional and surgical procedures:imaging findings and diagnostic performance study. J Ultrasound. 2020.https://pubmed.ncbi.nlm.nih.gov/32436181/.5. Parker R, Wuerdeman M, Grant M, Kitley C. Novel approach to uterineartery pseudoaneurysm embolization for delayed post-partum hemor-rhage. J Obstet Gynaecol Res. 2016;42(12):1870-1873.6. Eriksson L-G, Mulic-Lutvica A, Jangland L, Nyman R. Massive postpar-tum hemorrhage treated with transcatheter arterial embolization: tech-nical aspects and long-term effects on fertility and menstrual cycle.Acta Radiol. 2007;48(6):635-642.7. Kyeong K-S, Moon J-Y, Chae S-H, Hong SH, Kang M, Jeong EH. Del-ayed postpartum hemoperitoneum due to uterine artery pseudo-aneurysm rupture. Obstet Gynecol Sci. 2017;60(3):303-307.8. Dasari P, Maurya DK, Mascarenhas M. Uterine artery pseudo-aneurysm: a rare cause of secondary postpartum haemorrhage follow-ing caesarean section. Case Reports. 2011;2011:bcr0120113709.https://pubmed.ncbi.nlm.nih.gov/22700068/.How to cite this article: Pohlan J, Hinkson L, Wickmann U,Henrich W, Althoff CE. Pseudo aneurysm of the uterine arterywith arteriovenous fistula after cesarean section: A rare butsinister cause of delayed postpartum hemorrhage. J ClinUltrasound. 2021;49:265–268. https://doi.org/10.1002/jcu.22890268 POHLAN ET AL.

Pseudo aneurysm of the uterine artery with arteriovenous fistula after cesarean section: A rare but sinister cause of delayed postpartum hemorrhage

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Pseudo aneurysm of the uterine artery with arteriovenous fistula after cesarean section: A rare but sinister cause of delayed postpartum hemorrhage

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