Uterine necrosis following uterine artery embolization: case report and literature review

Introduction

A cesarean scar pregnancy (CSP) is a high-risk pregnancy that can result in uncontrollable bleeding, hysterorrhexis, peripheral visceral injury, and hysterectomy during or after curettage. Treatment includes medication, surgery, or a combination of these. CSP was divided into two types in 2000 by Vial et al.; however, this classification can not clearly guide clinical work (1). CSP was also classified into three types (I, II, III) according to the location and direction of growth of the gestational sac and myometrial thickness (2,3). It is much easier to select appropriate treatments using this classification (2,3). Uterine artery embolization (UAE) is an important adjuvant therapy that can be performed for CSP types II and III to decrease the risk of bleeding (3). The most common symptoms following UAE include abdominal pain, fever, and vaginal discharge. Reports have also documented episodes of endometrial, myometrial, and ovarian injuries, which can result in amenorrhea, ovarian failure, and impaired reproductive ability (4,5). Uterine necrosis is a major complication of embolization; however, it is extremely rare today. Uterine necrosis has a significant effect on the menstrual cycle, fertility, and subsequent pregnancies. Herein, we have presented a case of uterine necrosis after UAE for a CSP and have conducted a review of the literature available on PubMed. Only articles written in English were examined in this study. Our literature search did not identify any reports of uterine necrosis following UAE for a CSP. We present the following case in accordance with the CARE reporting checklist (available at https://gpm.amegroups.com/article/view/10.21037/gpm-21-46/rc).

Case presentation

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal. A 43-year-old woman was admitted to a local hospital and diagnosed with type III CSP and adenomyosis. She had a history of one cesarean section. Her serum human chorionic gonadotropin level was >200,000 mIU/mL. Prior to UAE, magnetic resonance imaging (MRI) revealed a gestational sac along the cesarean scar (Figure 1). The patient underwent curettage after UAE. Bilateral UAE was performed using polyvinyl alcohol embolization microsphere (100−300 µm and 300−500 µm) and gelatin sponge particles (560−710 µm). Three months after UAE, the patient was admitted to our hospital due to an asymptomatic vaginal mass (symptoms such as abdominal pain, abnormal vaginal bleeding, or fever were absent). On physical examination, a large, soft vaginal mass and a large uterus corresponding to the size of a four-month-old pregnancy, were detected. The cervix was not assessed at this time. MRI revealed a large mass occupying the uterine cavity, cervical canal, and upper portion of the vagina. The mass was continuous with the myometrium of the fundus uteri and exhibited traction on the fundus, which resulted in incomplete uterine inversion (Figure 2). The patient underwent an exploratory laparotomy, and a hysterectomy and bilateral salpingectomy were performed. Intraoperatively, extensive adhesions were noted in the pelvic cavity. Majority of the myometrium was necrotic, and the right side of the fundus was concave. A large, light-yellow mass was noted originating from the fundus and extending into the uterine cavity and vagina (Figures 3,4). Histopathological examination of the resected specimen showed extensive necrosis, as well as blood and inflammatory cell infiltration in the myometrium. Gelatin sponge components were also noted in the blood vessels of the myometrium. The exploratory laparotomy was completed in 2 hours, and approximately 200mL of blood was lost intraoperatively. The patient recovered well from surgery, and no complications were noted over the 2-year follow-up period.

Figure 1 MRI demonstrating a cesarean scar pregnancy and adenomyosis. MRI, magnetic resonance imaging.

Figure 2 MRI demonstrating a large mass and incomplete uterine inversion. MRI, magnetic resonance imaging.

Figure 3 The uterus and mass.

Figure 4 Extensive necrosis of the myometrium.

Discussion

UAE is an important adjuvant therapy for CSP; it blocks the blood flow to the gestational sac, which lowers the risk of intraoperative bleeding and a hysterectomy during or after uterine curettage (6). Among 841 patients who underwent UAE prior to uterine curettage, good hemostasis was achieved in 93.7% (7). Another retrospective study involving 169 patients with CSP who underwent UAE, demonstrated a 96.4% hemostasis success rate during dilation and curettage. UAE is also associated with a reduction in menstrual blood volume and future pregnancy rate (8).

Uterine necrosis is a rare complication of UAE and remains an important issue in the diagnosis and management of this condition. We conducted a search of English articles on PubMed and found 21 reports of uterine necrosis following UAE (9-26). The search strings were the following: uterine necrosis and uterine artery embolization. The clinical characteristics and outcomes of the patients in these studies are listed in Table 1. UAE was performed for three cases of leiomyoma and 18 cases of postpartum hemorrhage (PPH). In all 21 cases, uterine necrosis was managed with embolization agents, such as the Spongostan gelfoam slurry (Gelfoam) (n=9,42.9%), polyvinyl alcohol particle (PVA) (n=5, 23.8%), absorbable gelatin sponge (Curaspon) (n=6, 28.6%), and gelatin sponge pledges (Gelatin) (n=2,0.1%). The most common clinical symptoms after UAE were fever, abdominal pain, and vaginal discharge. The interval between UAE and the diagnosis of uterine necrosis ranged from 4–69 days. Uterine necrosis was often initially demonstrated by ultrasound and later confirmed by computed tomography (CT) and MRI. The CT and MRI findings of uterine necrosis sometimes include uterine elongation, myometrial gas, peripheral contrast uptake, and absent myometrial contrast uptake (27). However, myometrial gas and peripheral contrast uptake can be normal after UAE and are not specific signs of necrosis. Our patient demonstrated extensive uterine necrosis following UAE, resulting in uterine inversion. She was diagnosed by MRI and underwent a hysterectomy for severe uterine necrosis.

Previous studies have identified possible risk factors for uterine necrosis following UAE (27,28). These include the embolization technique, size of the particles of the embolic agents used, absence of vascular anastomoses, and presence of sepsis (27). The incidence of uterine necrosis can be reduced in several ways. First, a suitable embolic agent should be chosen. The gelatin sponge is suitable for UAE because it has large particles, which are unlikely to occlude small arteries. Gelatin sponge particles in UAE should exceed 500 µm in diameter (28). The small particles were used in our patient and may have been one of the causes of uterine necrosis. Second, the speed at which the embolic agent is injected should be controlled. Faster injection may reroute the agent to distal anastomotic channels and result in embolization of vessels supplying surrounding tissues, such as those of the ovaries (27). Third, significant adenomyosis in our patient is a confounding factor because embolization (to prevent hemorrhage) before curettage also treats adenomyosis, which will necrose and be eliminated via the vaginal tract. Furthermore, the treatment of uterine necrosis should be individualized. This report has demonstrated that partial necrosis may be treated with hysteroscopic or laparoscopic excision, which preserves fertility, whereas large necrotic areas require subtotal or total hysterectomy, which sacrifices fertility.

In conclusion, uterine necrosis is a rare complication of UAE. Therefore, UAE must be practiced with caution, using large polyvinyl alcohol particles (>500 µm) and/or gelatin sponge >500 µm. Physicians should strictly control the indications for UAE and master the technique explicitly.

Acknowledgments

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://gpm.amegroups.com/article/view/10.21037/gpm-21-46/rc

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://gpm.amegroups.com/article/view/10.21037/gpm-21-46/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). The study was approved by Ethics Committee of the West China Second University Hospital and written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Vial Y, Petignat P, Hohlfeld P. Pregnancy in a cesarean scar. Ultrasound Obstet Gynecol 2000;16:592-3. [Crossref] [PubMed]
2. Zhang H, Huang J, Wu X, et al. Clinical classification and treatment of cesarean scar pregnancy. J Obstet Gynaecol Res 2017;43:653-61. [Crossref] [PubMed]
3. The Group of Family Planning, Society of Gynaecology and Obstetrics, Chinese Medical Association. Expert consensus of cesarean scar pregnancy. Chinese Journal of Obstetrics and Gynecology 2016;8:568-72.
4. Cheong JY, Kong TW, Son JH, et al. Outcome of pelvic arterial embolization for postpartum hemorrhage: A retrospective review of 117 cases. Obstet Gynecol Sci 2014;57:17-27. [Crossref] [PubMed]
5. Spies JB. The EMMY trial of uterine artery embolization for the treatment of symptomatic uterine fibroid tumors: randomized, yes, but a flawed trial nonetheless. J Vasc Interv Radiol 2006;17:413-5. [Crossref] [PubMed]
6. Ma Y, Yang C, Shao X. Efficacy comparison of transcatheter arterial embolization with gelatin sponge and polyvinyl alcohol particles for the management of cesarean scar pregnancy and follow-up study. J Obstet Gynaecol Res 2017;43:682-8. [Crossref] [PubMed]
7. Li SZ, Tian HA, Jia WW, et al. Effect and impact factors of prophylactic uterine artery embolization for reducing bleeding during uterine curettage in treatment of cesarean scar pregnancy. Chinese Journal of Interventional Imaging and Therapy 2020;17:157-61.
8. Li X, Niu H, Li J, et al. Clinical assessment of uterine artery embolization combined with curettage when treating patients with cesarean scar pregnancy: A retrospective study of 169 cases. J Obstet Gynaecol Res 2020;46:1110-6. [Crossref] [PubMed]
9. Ruiz Sánchez E, Peinado Rodenas J, Gil Martínez-Acacio L, et al. Uterine necrosis. A rare complication of embolisation due to post-partum haemorrhage. J Gynecol Obstet Hum Reprod 2021;50:101773. [Crossref] [PubMed]
10. Mutiso SK, Oindi FM, Hacking N, et al. Uterine Necrosis after Uterine Artery Embolization for Symptomatic Fibroids. Case Rep Obstet Gynecol 2018;2018:9621741. [Crossref] [PubMed]
11. Tanaka K, Muraoka Y, Toda Y, et al. Finegoldia magna myometritis with uterine necrosis after uterine artery embolisation. J Obstet Gynaecol 2017;37:688-9. [Crossref] [PubMed]
12. Jean dit Gautier E, Midulla M, Lucot JP, et al. Hyperbaric oxygen therapy in a case of vaginal and uterine necrosis after embolization for postpartum hemorrhage. Int J Gynaecol Obstet 2015;130:286-7.
13. Kwon JH. Uterine necrosis and hysterectomy in a postpartum hemorrhage patient who underwent repeated uterine artery embolization. Taiwan J Obstet Gynecol 2015;54:791-2. [Crossref] [PubMed]
14. Rohilla M, Singh P, Kaur J, et al. Uterine necrosis and lumbosacral-plexopathy following pelvic vessel embolization for postpartum haemorrhage: report of two cases and review of literature. Arch Gynecol Obstet 2014;290:819-23. [Crossref] [PubMed]
15. Bouvier A, Sentilhes L, Thouveny F, et al. Planned caesarean in the interventional radiology cath lab to enable immediate uterine artery embolization for the conservative treatment of placenta accreta. Clin Radiol 2012;67:1089-94. [Crossref] [PubMed]
16. Tseng JJ, Ho JY, Wen MC, et al. Uterine necrosis associated with acute suppurative myometritis after angiographic selective embolization for refractory postpartum hemorrhage. Am J Obstet Gynecol 2011;204:e4-6. [Crossref] [PubMed]
17. Sentilhes L, Ambroselli C, Kayem G, et al. Maternal outcome after conservative treatment of placenta accreta. Obstet Gynecol 2010;115:526-34. [Crossref] [PubMed]
18. Coulange L, Butori N, Loffroy R, et al. Uterine necrosis following selective embolization for postpartum hemorrhage using absorbable material. Acta Obstet Gynecol Scand 2009;88:238-40. [Crossref] [PubMed]
19. Kirby JM, Kachura JR, Rajan DK, et al. Arterial embolization for primary postpartum hemorrhage. J Vasc Interv Radiol 2009;20:1036-45. [Crossref] [PubMed]
20. Courbiere B, Jauffret C, Provansal M, et al. Failure of conservative management in postpartum haemorrhage: uterine necrosis and hysterectomy after angiographic selective embolization with gelfoam. Eur J Obstet Gynecol Reprod Biol 2008;140:291-3. [Crossref] [PubMed]
21. La Folie T, Vidal V, Mehanna M, et al. Results of endovascular treatment in cases of abnormal placentation with post-partum hemorrhage. J Obstet Gynaecol Res 2007;33:624-30. [Crossref] [PubMed]
22. Chitrit Y, Zafy S, Pelage JP, et al. Amenorrhea due to partial uterine necrosis after uterine artery embolization for control of refractory postpartum hemorrhage. Eur J Obstet Gynecol Reprod Biol 2006;127:140-2. [Crossref] [PubMed]
23. Porcu G, Roger V, Jacquier A, et al. Uterus and bladder necrosis after uterine artery embolisation for postpartum haemorrhage. BJOG 2005;112:122-3. [Crossref] [PubMed]
24. Torigian DA, Siegelman ES, Terhune KP, et al. MRI of uterine necrosis after uterine artery embolization for treatment of uterine leiomyomata. AJR Am J Roentgenol 2005;184:555-9. [Crossref] [PubMed]
25. Pirard C, Squifflet J, Gilles A, et al. Uterine necrosis and sepsis after vascular embolization and surgical ligation in a patient with postpartum hemorrhage. Fertil Steril 2002;78:412-3. [Crossref] [PubMed]
26. Godfrey CD, Zbella EA. Uterine necrosis after uterine artery embolization for leiomyoma. Obstet Gynecol 2001;98:950-2. [PubMed]
27. Poujade O, Ceccaldi PF, Davitian C, et al. Uterine necrosis following pelvic arterial embolization for post-partum hemorrhage: review of the literature. Eur J Obstet Gynecol Reprod Biol 2013;170:309-14. [Crossref] [PubMed]
28. Song YG, Woo YJ, Kim CW. Uterine artery embolization using progressively larger calibrated gelatin sponge particles. Minim Invasive Ther Allied Technol 2016;25:35-42. [Crossref] [PubMed]