Surgical uterus-preserving therapy after uterine rupture and haemorrhage due to placenta percreta in the first trimester following embolisation of the uterine arteries and B-Lynch suture in a previous pregnancy.

A woman in her early 30s in the 11 2/7 week of pregnancy was admitted with severe abdominal pain and emesis. One year prior, the patient had undergone hysteroscopic adhesiolysis to treat Asherman syndrome resulting from a prior pregnancy. Examination of the patient revealed a haemoperitoneum and an intact intrauterine pregnancy. Laparoscopic adhesiolysis and haemostasis was performed and the patient was transferred to the intensive care unit. Subsequent examination due to persistent abdominal pain revealed an occult iatrogenic perforation of the uterus and placenta percreta with spontaneous uterine rupture. Although treatment for placenta percreta has generally been hysterectomy, in this case, the rupture and perforation sites were resected, representing successful fertility preserving management for this oft-overlooked pregnancy complication.

Obstetrics and gynaecology, Pregnancy, Surgery, Emergency medicine, Ultrasonography

Spontaneous uterine rupture mainly occurs during the second or third trimester. The occurrence in the first trimester, as in our case, is extremely rare. Based on our review of the medical literature, in most of the similar reported cases of placenta percreta and haemorrhage, a total abdominal hysterectomy was performed.1 It is very likely that the rates of spontaneous uterine rupture in all trimesters will increase with the increasing prevalence of abnormally invasive placenta (AIP) in the last decades. Here, we report a case of successful surgical uterus-preserving therapy. Case presentation A pregnant woman (gravida 2, para 1) in her early 30s in the 11 2/7 week of gestation (based on last menstrual period) was admitted to our clinic by ambulance due to acute severe lower abdominal pain with emesis. The patient had a medical history of a secondary caesarean section 6 years prior with uterine atony requiring embolisation of the uterine arteries and B-Lynch sutures. She subsequently developed secondary sterility due to Asherman syndrome, which was treated via hysteroscopic adhesiolysis 5 years later. After this intervention, she conceived spontaneously. For optimisation of pregnancy outcome, she was prescribed acetylsalicylic acid and enoxaparin. During physical examination at our emergency department, the patient became increasingly haemodynamically unstable. Sonography revealed a haematoperitoneum and an intact intrauterine pregnancy of 12 weeks’ gestation. Laboratory analysis showed a haemoglobin level of 68 g/L. Investigations Diagnostic laparoscopy confirmed the haemoperitoneum with 3 L of blood in the abdomen. Adhesions from the posterior and anterior walls of the uterus were also found. After adhesiolysis, there was light bleeding from the posterior wall of the uterus and iatrogenic heavy bleeding from the uterine fundus. After compression and electrocoagulation of these sites, haemostasis was achieved. During the surgery, the patient received two units of packed red blood cells, 2 g tranexamic acid and 6 g fibrinogen. Postoperatively, she was transferred to the intensive care unit. The following day the patient was successfully extubated. She had persistent abdominal pain. Initial postoperative ultrasound examination showed no free fluid in the peritoneal cavity and a still intact intrauterine pregnancy. MRI revealed an occult iatrogenic perforation at the uterine fundus, and placenta percreta at the posterior wall of the uterus with suspected uterine rupture (figure 1), which was subsequently confirmed via ultrasound. Differential diagnosis Haemoperitoneum in early pregnancy can be caused by a bleeding corpus luteum cyst. No cyst was initially seen on ultrasound examination; however, the cyst could have ruptured previously. Another cause could be heterotopic pregnancy, another rare diagnosis; we found no sonographic indices for this. A bleeding endometriosis implant is another potential differential diagnosis for spontaneous haemoperitoneum in pregnancy. During the previous caesarean section, no evidence of endometriosis was documented.2 Treatment Relaparoscopy confirmed the iatrogenic perforation after electrocoagulation at that site in the first laparoscopy with prolapsing amniotic sac at the fundus (figure 2). There were also traces of electrocoagulation at the posterior wall of the uterus at the described site of placenta percreta with uterine rupture (figure 3). The iatrogenic perforation site was repaired via V-Loc sutures. The internal iliac arteries were temporarily occluded via bulldog clamps. The medically indicated termination of pregnancy via suction curettage was difficult due heavy vaginal bleeding and non-removable placenta. The ruptured placenta percreta site at the posterior wall of the uterus was resected with cold scissors and the pregnancy was retrieved intraabdominally. The incision was closed via continuous V-Loc sutures. Due to persistent heavy bleeding from the resection site, we converted to Pfannenstiel laparotomy. The bleeding site was sutured via layered repair with haemostatic patch for haemostasis and compression. Outcome and follow-up The recovery of the patient was uneventful, and she was discharged on postoperative day 5. Histology confirmed placenta percreta. We conducted weekly beta human chorionic gonadotropin testing to 0 IU/L. We recommended contraception for 1 year, and hydrosonography and MRI after 1 year to evaluate the uterine scar.

Placenta percreta-induced uterine rupture in the first trimester is extremely rare. There are only a few cases reported in the literature.3 4 It is very likely that the rate will increase with the increasing prevalence of AIP in the last decades. It has been reported that the incidence steadily increased from 1:25 000 in the 1950s to 1:2500 in the 1980s.5 6 A 20-year analysis in 2005 revealed an incidence of AIP of 1:533 in the USA.7 The trend continued and the recently reported numbers show an incidence of AIP of 1:272 in the USA.8 An AIP is not commonly diagnosed in the first trimester. Most are unfortunately retrospective diagnoses. Sonography or MRI can detect abnormal placentation, though sonography is more practical. Typical signs include loss of the ‘clear zone’, abnormal placental lacunae, bladder wall interruption, myometrial thinning, placental bulge, focal exophytic mass and several signs in two-dimensional colour Doppler such as bridging vessels.9 Most AIPs are diagnosed in the second or third trimester of pregnancy. There are reports that suggest that a diagnosis of AIP is possible in the first trimester with sonography. In the first trimester, the signs for AIP are a low implantation of the gestational sac close to a previous uterine scar, anechoic spaces within the placental mass (lacunae) and reduced myometrial thickness.10 In our case, there were no sonographic indicators of AIP found on initial emergency examination. After the MRI and repeated sonography, reduced myometrial thickness penetrating the uterine serosa, loss of the ‘clear zone’, and bridging vessels were observed, clearly indicating placenta percreta. The haemoperitoneum can be attributed to uterine rupture at the site of the placenta percreta. Predisposing factors for abnormal placentation include prior uterine surgery, especially with entrance to the uterine cavity; previous caesarean section; short interval between successive caesarean sections; increased maternal age; in vitro fertilisation and smoking.5 7 Risk factors in our patient were previous caesarean section and hysteroscopic adhesiolysis with possible occult perforation. This could explain the localisation of the placenta percreta at the posterior wall of the uterus rather than the caesarean scar. Uterine perforation is possible during any gynaecological procedure involving instrumentation of the uterus.11 The reported risk of perforation during a hysteroscopic procedure is 1.61% and adhesiolysis has a significantly higher risk than other indications.12 Due to the unusual location of the patient’s placenta percreta, it was not initially detected, and this catastrophic uterine rupture could have been overlooked. Therefore, the possibility of AIP should be considered and managed urgently in symptomatic patients with high risk of adherent placenta. Uterine perforation during laparoscopy in pregnancy is a rare complication, and in our case was most likely iatrogenic. The myometrium of the gravid uterus is softer and more vulnerable to perforation.13 We suspect that electrocoagulation, used to stop the bleeding from the adhesiolysis, was the cause of the perforation.14 We caution against any coagulation of the gravid uterus, and recommend suture closure if significant bleeding or injury of the uterus is observed. Nevertheless, laparoscopy during pregnancy appears to be a safe and effective surgical approach.15 In this case, the patient had previously undergone embolisation of the uterine arteries due to postpartum haemorrhage (PPH). We reviewed the literature for subsequent pregnancy outcome, in this case uterine rupture, after embolisation for PPH. Soyama et al reported a case with uterine rupture after embolisation for PPH; however, in their review of the literature they found no significant correlation between uterine rupture and previous embolisation. Interestingly two women of the collective (n=94) suffered from placenta accreta and required hysterectomy.16 The previous therapeutic choice for placenta percreta was hysterectomy. Procedures to preserve fertility were considered to be of an experimental nature.1 Nevertheless, Wang et al reported a uterus preserving case of laparoscopic management of placenta increta.17 Further therapeutic options, if fertility or uterine preservation is desired, are uterine artery embolisation, cytotoxic therapy with methotrexate or hysteroscopy with lesion resection.17 18 There are other reported cases of uterus preserving management of uterine rupture during pregnancy, but not due to AIP.11 19–23 We found two other cases of placenta percreta-induced uterine rupture in the first trimester (defined according to Swiss standard as first day of LMP to 12 0/7 weeks). In one case, the placenta percreta was at the site of the prior caesarean section scar and a total abdominal hysterectomy was performed, and in the other case with no prior caesarean section a uterine reconstruction was performed with layer-by-layer suture.3 4 Hou et al also performed hysterorrhaphy after uterine rupture caused by placenta percreta, but in the second trimester of gestation.24 In conclusion, this case represents a very rare, fertility preserving clinical management after placenta percreta-induced uterine rupture in early pregnancy. This report highlights the fact that uterine rupture and AIP should be considered in the differential diagnosis of all pregnant women who present with acute abdomen, even in the first trimester. Further investigation of the acute abdomen with laparoscopy is recommended, being careful to avoid coagulation on the gravid uterus. Patient’s perspective. ‘I am very relieved that I was able to keep my uterus and that I still have the option to get pregnant again, as I may still want children in the future. I recovered well from the operations and hospitalisation; however my husband found the critical nature of the situation challenging.’ (Translated from German with permission from the patient) Learning points. Uterine rupture and abnormally invasive placenta (AIP) should be considered in the differential diagnosis of all pregnant women who present with acute abdomen. Hysteroscopic adhesiolysis is associated with a significantly higher risk for uterus perforation than other indications, and a perforation can lead to serious complications in a subsequent pregnancy, as in our case. Laparoscopy is safe in pregnancy and very useful in the diagnostic of an acute abdomen. Avoid coagulation on the gravid uterus; it could lead to perforation. Laparoscopic uterine preserving therapy is a viable alternative to hysterectomy in cases of AIP in the first trimester. Acknowledgments The authors would like to thank Susanne Forst for her support in finalising this article. Footnotes Contributors: LG: conception and design, analysis and interpretation of data, drafting the article, final approval. LE: analysis and interpretation of data, revising it critically for important intellectual content drafting the article, final approval. UF: acquisition, revising it critically for important intellectual content, final approval. GM: conception and design, acquisition, revising it critically for important intellectual content, final approval. Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors. Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy. Competing interests: None declared. Provenance and peer review: Not commissioned; externally peer reviewed. Ethics statements Patient consent for publication Consent obtained directly from patient(s)

- 1.Höpker M, Fleckenstein G, Heyl W, et al. Placenta percreta in week 10 of pregnancy with consecutive hysterectomy: case report. Hum Reprod 2002;17:817–20. 10.1093/humrep/17.3.817 [DOI] [PubMed] [Google Scholar] - 2.Cozzolino M, Corioni S, Maggio L, et al. Endometriosis-Related hemoperitoneum in pregnancy: a diagnosis to keep in mind. Ochsner J 2015;15:262–4. [PMC free article] [PubMed] [Google Scholar] - 3.Dabulis SA, McGuirk TD. An unusual case of hemoperitoneum: uterine rupture at 9 weeks gestational age. J Emerg Med 2007;33:285–7. 10.1016/j.jemermed.2007.01.009 [DOI] [PubMed] [Google Scholar] - 4.Cho MK, Ryu HK, Kim CH. Placenta Percreta-Induced Uterine Rupture at 7th Week of Pregnancy After In Vitro Fertilization in a Primigravida Woman: Case Report. J Emerg Med 2017;53:126–9. 10.1016/j.jemermed.2017.01.041 [DOI] [PubMed] [Google Scholar] - 5.Miller DA, Chollet JA, Goodwin TM. Clinical risk factors for placenta previa-placenta accreta. Am J Obstet Gynecol 1997;177:210–4. 10.1016/S0002-9378(97)70463-0 [DOI] [PubMed] [Google Scholar] - 6.Committee on Obstetric Practice . Committee opinion no. 529: placenta accreta. Obstet Gynecol 2012;120:207–11. 10.1097/AOG.0b013e318262e340 [DOI] [PubMed] [Google Scholar] - 7.Wu S, Kocherginsky M, Hibbard JU. Abnormal placentation: twenty-year analysis. Am J Obstet Gynecol 2005;192:1458–61. 10.1016/j.ajog.2004.12.074 [DOI] [PubMed] [Google Scholar] - 8.Mogos MF, Salemi JL, Ashley M, et al. Recent trends in placenta accreta in the United States and its impact on maternal-fetal morbidity and healthcare-associated costs, 1998-2011. J Matern Fetal Neonatal Med 2016;29:1077e82:1077–82. 10.3109/14767058.2015.1034103 [DOI] [PubMed] [Google Scholar] - 9.Collins SL, Ashcroft A, Braun T, et al. Proposal for standardized ultrasound descriptors of abnormally invasive placenta (AIP). Ultrasound Obstet Gynecol 2016;47:271–5. 10.1002/uog.14952 [DOI] [PubMed] [Google Scholar] - 10.D'Antonio F, Timor-Tritsch IE, Palacios-Jaraquemada J, et al. First-Trimester detection of abnormally invasive placenta in high-risk women: systematic review and meta-analysis. Ultrasound Obstet Gynecol 2018;51:176–83. 10.1002/uog.18840 [DOI] [PubMed] [Google Scholar] - 11.Amro B, Lotfi G. Spontaneous rupture of an unscarred uterus in early pregnancy: a rare but life-threatening emergency. BMJ Case Rep 2019;12:e228493. 10.1136/bcr-2018-228493 [DOI] [PMC free article] [PubMed] [Google Scholar] - 12.Agostini A, Cravello L, Bretelle F, et al. Risk of uterine perforation during hysteroscopic surgery. J Am Assoc Gynecol Laparosc 2002;9:264–7. 10.1016/S1074-3804(05)60401-X [DOI] [PubMed] [Google Scholar] - 13.Rowlands S, Oloto E, Horwell DH. Intrauterine devices and risk of uterine perforation: current perspectives. Open Access J Contracept 2016;7:19–32. 10.2147/OAJC.S85546 [DOI] [PMC free article] [PubMed] [Google Scholar] - 14.Syam HH. Pregnancy outcomes following laparoscopic myomectomy. WJOLS 2009;1:35–40. 10.5005/jp-journals-10007-1045 [DOI] [Google Scholar] - 15.Buser KB. Laparoscopic surgery in the pregnant patient: results and recommendations. JSLS 2009;13:32–5. [PMC free article] [PubMed] [Google Scholar] - 16.Soyama H, Miyamoto M, Sasa H, et al. Pregnancy with asymptomatic uterine complete rupture after uterine artery embolization for postpartum hemorrhage. Taiwan J Obstet Gynecol 2017;56:538–40. 10.1016/j.tjog.2016.08.008 [DOI] [PubMed] [Google Scholar] - 17.Wang Y-L, Su T-H, Huang W-C, et al. Laparoscopic management of placenta increta after late first-trimester dilation and evacuation manifesting as an unusual uterine mass. J Minim Invasive Gynecol 2011;18:250–3. 10.1016/j.jmig.2010.11.001 [DOI] [PubMed] [Google Scholar] - 18.Farahnaz F, Arezoo E. Placenta accreta at 15 weeks of gestational age and uterus preservation with hysterectomy abortion. Zahedan J Res Med Sci 2017;19:e10830. [Google Scholar] - 19.Sun H-D, Su W-H, Chang W-H, et al. Rupture of a pregnant unscarred uterus in an early secondary trimester: a case report and brief review. J Obstet Gynaecol Res 2012;38:442–5. 10.1111/j.1447-0756.2011.01723.x [DOI] [PubMed] [Google Scholar] - 20.Wang L-M, Wang P-H, Chen C-long, et al. Uterine preservation in a woman with spontaneous uterine rupture secondary to placenta percreta on the posterior wall: a case report. J Obstet Gynecol Res 2009;35:379–84. 10.1111/j.1447-0756.2008.00936.x [DOI] [PubMed] [Google Scholar] - 21.Park Y-J, Ryu K-Y, Lee J-I, et al. Spontaneous uterine rupture in the first trimester: a case report. J Korean Med Sci 2005;20:1079–81. 10.3346/jkms.2005.20.6.1079 [DOI] [PMC free article] [PubMed] [Google Scholar] - 22.Takahashi T, Ota K, Jimbo M, et al. Spontaneous unscarred uterine rupture and surgical repair at 11 weeks of gestation in a twin pregnancy. J Obstet Gynaecol Res 2020;46:1911–5. 10.1111/jog.14396 [DOI] [PubMed] [Google Scholar] - 23.Dos Anjos Siqueira I, Murugappan S, Howat P, et al. Posterior uterine rupture in early first trimester. BMJ Case Rep 2021;14:e244801. 10.1136/bcr-2021-244801 [DOI] [PMC free article] [PubMed] [Google Scholar] - 24.Hou J-H, Lee T-H, Wang S-Y, et al. Spontaneous uterine rupture at a non-cesarean section scar site caused by placenta percreta in the early second trimester of gestation: a case report. Taiwan J Obstet Gynecol 2021;60:784–6. 10.1016/j.tjog.2021.05.037 [DOI] [PubMed] [Google Scholar]