Multidisciplinary team management of caesarean scar ectopic pregnancy progressing to a live birth and caesarean hysterectomy at 34 weeks: A case report

Abstract

Progression of a caesarean scar ectopic pregnancy (CSEP) to a live birth is exceptionally rare. Whether the placenta should be removed during a caesarean section for patients with a CSEP complicated by severe placenta accreta spectrum remains unclear. This report presents the case of a 42-year-old multigravida with two prior caesarean sections who presented with CSEP at 6 weeks. Despite recommendations for termination, the patient decided to continue the pregnancy. Serial imaging confirmed a progressive placenta accreta spectrum. At 34+ weeks of gestation, a caesarean hysterectomy was successfully performed under the management of a multidisciplinary team, with good maternal and infant outcomes. The management of a CSEP progressing to a live birth during the third trimester requires provider expertise and multidisciplinary treatment and should be individualized. In the present case, caesarean hysterectomy was performed without attempting placental removal, which might have significantly decreased blood loss. While the patient survived, the management remains controversial, and women with CSEP opting for expectant management should be informed about the lack of conclusive evidence on its safety and associated risks. Such options can be considered only in a highly equipped specialist centre with access to a multidisciplinary team.

Highlights

• •Progression of a caesarean scar ectopic pregnancy (CSEP) to a live birth is exceptionally rare.
• •It is unclear whether the placenta should be removed during a caesarean section for patients with CSEP complicated by severe placenta accreta spectrum.
• •The management of CSEP should be individualized and requires provider expertise and multidisciplinary input.
• •A strategy of caesarean hysterectomy without attempting placental removal, might have significantly decreased blood loss.

1. Introduction

Caesarean scar ectopic pregnancy (CSEP), also referred to as caesarean scar pregnancy (CSP), is a rare type of uterine ectopic pregnancy [1,2] in which the pregnancy implants into a myometrial defect caused by a caesarean scar. Its incidence is predicted to increase given the global increase in the rates of caesarean section. An analysis of world data for the period 2010–2018 and from 154 countries, covering 94.5 % of global live births, revealed that 21.1 % of pregnant women gave birth by caesarean section (CS) [2]. It has been estimated that by 2030, 28.5 % of women worldwide will be delivered by CS, with the greatest increase predicted to be in East Asia.

The recommendation is that cervical, caesarean scar, and intramural ectopic pregnancies be considered distinct types of uterine ectopic pregnancies [3]. Given that most CSEPs present as failed pregnancies or patients choose pregnancy termination, data on their natural history are limited [4]. Maintenance of the pregnancy is a high-risk option for both mother and child, while clinicians and medical institutions encounter challenges in providing safe and effective care for these patients.

2. Case Presentation

A 42-year-old pregnant woman presented to the tertiary care centre with a CSEP at 6+ weeks of gestation. She had a history of two caesarean sections and three ectopic pregnancies. This was a natural pregnancy. In early pregnancy, ultrasound at a local hospital revealed a gestational sac of 18 × 12 × 7 mm in the lower segment of the uterine cavity, with a wedge-shaped protrusion into the incision site of the anterior wall muscle layer. A 4 × 2 mm embryo echo was observed inside, with primitive heartbeats. No symptoms of abdominal or vaginal bleeding were observed. The local hospital suggested terminating the pregnancy but the patient refused and visited the tertiary care obstetrics department.

Ultrasound revealed that the embryo was approximately 11 mm long and had primitive heartbeats. The lower segment of the uterine isthmus embryo sac penetrated the anterior muscular layer and was 3.0 mm from the uterine serosal layer. Doppler blood flow signals were observed anterior to the embryo sac. Based on the ultrasound findings, a diagnosis of CSEP was made (Fig. 1), and the patient was admitted to the gynaecology ward.

Fig. 1.

The gestational sac implanted into a myometrial defect caused by a caesarean scar during early pregnancy is shown.

Magnetic resonance imaging (MRI) revealed that the embryo sac was in the lower segment of the uterine cavity and partially protruded towards the anterior wall incision. Again, termination of the pregnancy was suggested, but the patient continued to refuse this. A decision to continue the pregnancy was made after written acknowledgement of the risk of this pregnancy was provided. At 11+ weeks of gestation, obstetric outpatient ultrasound revealed that the NT was normal, but the lower edge of the placenta covered the cervical inner opening.

At 12 weeks of gestation, the patient again sought medical attention and requested the establishment of an obstetrical process during an antenatal check-up. At 22+ weeks of gestation, prenatal ultrasound suggested that the position of the placenta extended from the anterior wall to the posterior wall and covered the cervical inner opening, with abundant Doppler blood flow signals in the anterior isthmus muscle layer. At 26 weeks, ultrasound imaging suggested that the lower segment of the anterior muscle layer was thin, with unclear boundaries from the placenta. MRI suggested that the lower edge covered the cervical opening. Placental implantation and haematoma formation were considered on the basis of multiple abnormal signal foci in the placenta (Fig. S1).

Ultrasound examinations at 31+ and 33+ weeks of gestation indicated placental implantation, and abundant blood flow signals were observed between the placenta and uterine wall. At 34 weeks, MRI revealed that the placenta was located on the anterior wall, cervical opening, and posterior wall of the uterus, with the lower edge completely covering the cervical opening. The placental signal was uneven, with a scattered, small, patchy, high T2W signal. Complete placenta previa and placental implantation were considered. Uneven placental signals can be accompanied by scattered blood sinuses. The patient was admitted at approximately 35 weeks.

Preoperative discussions by the multidisciplinary team were organized, and surgical plans were formulated to ensure medical safety. Considering the risks associated with placenta previa and placental implantation, the multidisciplinary team reached a consensus that, owing to the high risk of postpartum haemorrhage during delivery, a planned caesarean hysterectomy with the protective use of abdominal aortic balloon occlusion would be performed; to decrease blood loss during surgery, removal of the placenta would not be attempted. The obstetrics safety office coordinated the efforts of various experts, including obstetricians, gynaecologists, anaesthesiologists, intensive care doctors, radiologists, nursing staff, laboratory doctors, blood banks, physicians, and ultrasound doctors, to ensure safety and security. The patient's family members were fully informed of the critical conditions and risks.

Under preset balloon occlusion of the abdominal aorta, the patient underwent a classical CS (a vertical incision in the upper uterine body) under general anaesthesia. After entering the abdominal cavity, the obstetrician conducted rapid exploration of the lower segment of the uterus to verify the preoperative judgement of “placental penetrative implantation” and reconfirmed the plan for caesarean hysterectomy. Before the uterus was opened, the abdominal aortic balloon was filled to temporarily reduce blood flow, and the obstetrician quickly delivered the foetus. After the umbilical cord was cut, the obstetrician tied the placental side of the umbilical cord with sutures, filled the uterine cavity with a large gauze without attempting to remove the placenta, and quickly sutured it to close the CS incision. A gynaecologist performed a total hysterectomy because of indications of “dangerous placenta previa and placental implantation” (Fig. 2a).

Fig. 2.

(a) and (b) The penetrating implantation of the placenta on the anterior wall of the lower uterine segment. As shown in Fig. 2a, placental implantation penetrated the uterine serosa layer. The uterine specimen is shown in Fig. 2b: the placenta had penetrated and merged into the lower segment of the uterus.

After the hysterectomy, a uterine specimen was cut for examination. This showed that the placenta was attached to the anterior wall of the lower segment of the uterus, completely covering the inner opening to the posterior wall, with a penetrating area of 8 × 6 cm of placental implantation on the anterior wall of the lower segment of the uterus (Fig. 2b).

The duration of surgery was 3+ hours. The intraoperative blood loss was 1690 ml, with 250 ml of autologous blood transfusion, 4 units of red blood cell suspension, 400 ml of plasma, 8 g of fibrinogen, 1200 units of prothrombin complex, 100 ml of aminomethylbenzoic acid, and 3000 ml of crystalloids.

After surgery, the patient was admitted to the intensive care unit and transferred to the general obstetric ward on the second day. Several days later, she was discharged; routine postpartum follow-up showed satisfactory recovery. A pathological examination revealed that the entire uterus opening was 26 cm × 17 cm × 7 cm, and the area of placental implantation and the lower segment of the uterus was approximately 15 × 11 cm. Partial placental tissue penetrated the left serosa.

3. Discussion

CSEP is a variant of uterine ectopic pregnancy according to the 2020 definition provided by the European Society of Human Reproduction and Embryology [3]. CSEP is closely associated with caesarean delivery and embryo implantation [5]. The continuous global increase in caesarean deliveries is causing a parallel increase in CSEP [6,7], although it is still rare, with an estimated incidence of 1/1800–1/2500 for all caesarean deliveries [8]. Transvaginal ultrasonography and colour Doppler are the most useful diagnostic tools [4]. CSEP is a precursor of placenta accreta spectrum (PAS) [9], which includes placenta accreta, placenta increta, and placenta percreta. This is a life-threatening condition often associated with serious obstetric complications, such as massive postpartum haemorrhage and hysterectomy [10,11].

The Society for Maternal-Foetal Medicine recommends against expectant management of CSEP [12], and few patients with CSEP decline to terminate their pregnancy. In the present case, the patient was repeatedly informed of the great risks of CSEP but refused termination, although she did agree to undergo a possible hysterectomy. Clinicians and medical institutions face great challenges, and intensive management must be maintained during the ongoing pregnancy and the caesarean section [13].

The treatment and management of CSEP require expertise, multidisciplinary management, and scientific decision-making regarding surgical plans and should be based on individual risk factors to prevent complications. For women who choose expectant management, the timing of caesarean delivery should be individualized but is typically between 34 and 36 weeks of gestation [12,14]. There is a previous report of a patient receiving expectant management of CSEP who successfully underwent caesarean delivery in the 38th week because of asymptomatic features [7]. A systematic review and meta-analysis of 17 studies on expectant management of CSEP published after 2000 reported that 40 patients (76.9 %) progressed to the third trimester of pregnancy, and hysterectomy during caesarean delivery was required in 60.6 % of cases [15].

For patients with placenta penetrative implantation and an extensive implantation area that has already invaded the uterus or cervix, hysterectomy is required. It must be made clear that such an option can be considered only in a highly equipped environment with access to a multidisciplinary team, blood products and an intensive care unit. The treatment of CSEP with PAS should ideally be performed at specialized tertiary centres, as the initial care provided at primary or secondary maternity care facilities is an important risk factor for undiagnosed CSEP [16]. An excellent multidisciplinary team can optimize maternal and neonatal outcomes with accreta. A caesarean section should be performed by an experienced practitioner and a senior anaesthetist, and other consultants should be available. An intensive care unit and a hospital transfusion laboratory capable of obtaining blood products immediately are needed. Allogeneic blood transfusions also play an important role during surgery.

The present case emphasizes the importance of a planned caesarean delivery without attempting to remove the placenta (which decreases blood loss) for a CSEP with a severe placenta accreta spectrum, followed by a planned hysterectomy [17]. Multidisciplinary input and approaches play key roles in the successful expectant management of CSEP complicated by severe placenta accreta spectrum. A recent systematic review revealed wide variation in outcomes and high-quality research is urgently needed [18]. The present report is intended to solely to share experience and there is no intention to encourage the routine use of expectant management of CSEP, which should be reserved for exceptional circumstances where a patient declines termination of the pregnancy.

4. Conclusion

In conclusion, the successful management of CSEP progressing to a live birth in the third trimester requires provider expertise and multidisciplinary treatment and should be individualized. The case illustrates caesarean hysterectomy without attempting placental removal in the setting of severe placenta accreta spectrum, which might significantly decrease blood loss during caesarean section. While the patient survived, the management remains controversial, and women opting for expectant management should be informed about the lack of conclusive evidence on its safety.

Contributors

Ruihong Xue contributed to patient care, acquisition and interpretation of the data, drafting of the manuscript, review of the literature, and critical revision of the manuscript for important intellectual content.

Wei Gu contributed to the operation and critical revision of the manuscript for important intellectual content.

Xiao Lang contributed to patient care, conception of the case report, and critical revision of the manuscript for important intellectual content.

All the authors approved the final submitted manuscript.

Patient consent

Written informed consent was obtained from the patient for the publication of this case report and the accompanying images.

Provenance and peer review

This article was not commissioned and was peer reviewed.

Funding

This work did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Declaration of competing interest

The authors declare that they have no competing interest regarding the publication of this case report.

Appendix A. Supplementary data

Supplementary material