Bilateral haemorrhagic cysts on ultrasound with an intraoperative diagnosis of spontaneous bilateral unruptured ectopic pregnancies: A case report with management challenges in a rural setting

Abstract

Spontaneous bilateral ectopic pregnancy is extremely rare, with an estimated incidence of 1 in 200,000 spontaneous pregnancies. Diagnosis is challenging, particularly in rural and remote settings where clinicians may have limited experience with rare presentations and reduced access to advanced imaging and specialist support. This report presents the case of a 36-year-old woman (gravida4, para 3) who presented to a rural hospital with vaginal bleeding and pelvic pain at 4 + 5 weeks of gestation. Serial quantitative beta-human chorionic gonadotropin levels increased from 962 IU/L to 6545 IU/L over 15 days. Transvaginal ultrasound demonstrated no intrauterine pregnancy, a right adnexal mass, and bilateral haemorrhagic ovarian cysts. She was admitted with suspected right tubal ectopic pregnancy. After discussion regarding the ultrasound findings and management options, the patient was consented for a right salpingectomy. However, at laparoscopy, bilateral unruptured tubal ectopic pregnancies were identified. Following an intraoperative discussion with next of kin, a right salpingectomy and additional left salpingostomy were performed to preserve natural fertility. Her postoperative recovery was uncomplicated and she was discharged home with serial quantitative testing of beta-human chorionic gonadotropin levels until negative. Histopathology confirmed bilateral tubal ectopic pregnancies. Bilateral ectopic pregnancy is difficult to diagnose preoperatively. This case highlights the potential diagnostic significance of bilateral corpus lutea and the importance of maintaining a high index of suspicion. In rural settings, limited access to sonographic expertise and fertility services may complicate both diagnosis and management. Bilateral ectopic pregnancy should be considered in suspected ectopic pregnancies with bilateral adnexal findings. Careful imaging review and fertility-conscious surgical planning are essential, particularly in rural and remote healthcare settings.

Highlights

• •Spontaneous bilateral tubal ectopic pregnancy is rare.
• •In the reported case, ultrasound demonstrated bilateral haemorrhagic ovarian cysts preoperatively.
• •Bilateral unruptured ectopic pregnancies were diagnosed at laparoscopy.
• •Bilateral adnexal findings may prompt earlier clinical suspicion.
• •Fertility-preserving surgery was performed in a rural hospital.

1. Introduction

The incidence of ectopic pregnancy in Australia is estimated at 2%, with 4 maternal deaths due to ruptured ectopic pregnancies between 2011 and 2020 [1], [2]. However, spontaneous bilateral ectopic pregnancy is extremely rare, with an incidence of 1:200000 spontaneous pregnancies and 1:725–1580 of all ectopic pregnancies [3], [4].

This report presents an intraoperative diagnosis of spontaneous bilateral unruptured tubal ectopic pregnancies in rural setting. Following discussion with next of kin, a right salpingectomy and left salpingostomy were performed. The case also demonstrates ultrasound evidence of bilateral ovarian cysts as a potential indicator of bilateral ectopic pregnancies.

2. Case Presentation

A 36-year-old woman (gravida 4, para 3) presented to a rural hospital with vaginal bleeding and cramping at 4 + 5 weeks of gestation. Her past medical history included anxiety, 3 previous lower-segment caesarean sections, 1 previous sexually transmitted infection (STI) that was appropriately treated and an open appendicectomy. She was a non-smoker. Quantitative testing found a beta-human chorionic gonadotropin (βHCG) level of 962 IU/L. Transvaginal ultrasound performed at the rural hospital showed no evidence of intrauterine pregnancy; however, a 1.3 × 1.2 × 0.9 cm ovoid structure was noted in the right ovary, considered most consistent with a corpus luteum. STI testing was negative.

The patient was discharged home with outpatient follow-up. She had had a subdermal etonogestrel implant inserted 2 weeks prior with a negative urine βHCG at that time. 5 days before presenting to hospital she tested positive on a urine βHCG and her subdermal implant was removed. A follow-up ultrasound, again performed at the same rural hospital 15 days later, demonstrated no evidence of an intrauterine pregnancy and trace free fluid in the canal. In her right adnexa there was a 1.5 × 1.3 × 1.3 cm hyperechoic mass with peripheral vascularity separate and lateral to the right ovary. It was tender with probe manipulation (Fig. 1). The right ovary contained a 3.3 cm haemorrhagic cyst and the left ovary contained a 2.8 cm likely haemorrhagic follicle (Fig. 1 & Fig. 2). There was no free fluid in the pouch of Douglas. She was admitted from radiology with a likely diagnosis of right tubal ectopic pregnancy with a βHCG level of 6545 IU/L and a haemoglobin level of 12.6 g/dL. Her management options were discussed and she was consented for a diagnostic laparoscopy +/− right salpingectomy +/− D&C +/− intra-uterine device insertion.

Fig. 1.

Right adnexa with hyperechoic vascular mass and ovarian haemorrhagic cyst.

Fig. 2.

Left adnexa with ovarian haemorrhagic follicle.

Intraoperatively she was found to have bilateral unruptured tubal ectopic pregnancies with a small haemoperitoneum (Fig. 3). The principal surgeon unscrubbed to discuss the findings with the next of kin. This included the options of bilateral salpingectomy with in-vitro fertilisation (IVF) required for future pregnancies or right salpingectomy and left salpingostomy with the risk of intraoperative bleeding or failure requiring salpingectomy. The decision was made for the fertility-preserving approach of a right salpingectomy and left salpingostomy only.

Fig. 3.

Bilateral unruptured tubal ectopic pregnancies.

The patient was debriefed by the principal surgeon postoperatively. She recovered well and was discharged home on day 1 postoperatively with outpatient follow-up. Her βHCG level appropriately down-trended in the following weeks and no further surgical interventions were required. Histopathology confirmed bilateral tubal ectopic pregnancies.

3. Discussion

Ectopic pregnancies represent 2% of all pregnancies in Australia [1]. At diagnosis 95% are tubal with 85% unruptured [5]. Risk factors include smoking, previous ectopic pregnancies, multiple sexual partners, previous STIs and/or pelvic inflammatory disease, infertility, previous tubal surgeries, tubal pathology, previous terminations, previous caesarean sections, microprogestins, and artificial reproductive technologies [6], [7].

The presence of a subdermal implant in this case is unlikely to have contributed to the location of the pregnancy. Guidelines recommend subdermal implant insertion on day 1–5 of the patient's menstrual cycle when pregnancy is excluded; however, if not within this period, they recommend barrier contraception use for 7 days [8]. An early pregnancy (less than three weeks) cannot be excluded with a negative urine βHCG [8]. Thus, the patient was likely pregnant prior to the subdermal implant insertion. This is in keeping with an Australian study in 2004 that found 21% of pregnancies with a subdermal implant in situ were due to pregnancies prior to insertion [9]. Additionally, a history of caesarean section, as in the present case, is only a risk factor for caesarean scar ectopic pregnancies [10].

There are three theorised causes of spontaneous bilateral ectopic pregnancy: simultaneous multiple ovulations, sequential impregnation, and transperitoneal migration of trophoblastic cells from one extra uterine pregnancy to the other tube with implantation [11]. There is a 5–10% chance of bilateral ovulation per menstrual cycle [12].

Diagnosis of ectopic pregnancy is based on quantitative βHCG trends and ultrasound findings [5]. Despite a rising incidence of spontaneous unilateral ectopic pregnancies due to the increasing use of early-pregnancy services and high-resolution transvaginal ultrasonography probes, bilateral ectopic pregnancies continue to be a diagnostic dilemma [1]. A review article in 2007, found only 2 of 42 bilateral ectopic pregnancies were diagnosed with ultrasound [3]. The accuracy of ultrasonography is directly related to the skills and experience of the sonographer. Transvaginal ultrasound has a sensitivity of 87–99% and specificity of 94–99% for unilateral ectopic pregnancy [13]. Despite this, even experienced sonographers will encounter only a few bilateral ectopic pregnancies throughout their career, making accurate identification difficult [14]. This limitation is unlikely to change without improving technology. In this case, bilateral ovarian cysts were present. These could represent two corpus luteums and provide evidence of simultaneous multiple ovulations or sequential impregnation. This was not recognised at the time. Studies have also shown that a corpus luteum can even be located on the contralateral side to the ectopic on ultrasound [15]. Furthermore, the presence of two or more corpus luteums should prompt more thorough evaluation for bilateral ectopic pregnancy. This is particularly important in rural or resource-limited settings, where access to specialist input may be delayed and clinicians may have fewer opportunities to gain experience with rare pathologies. Another suggested method of diagnosis is cell-free pregnancy-associated microRNAs in maternal plasma which have been found to be statistically significantly different in women with an ectopic pregnancy [16].

Management options on ectopic pregnancies include conservative monitoring, medical – with methotrexate – and surgical [5]. The safety and recommendation of these options is dependent on the location and size of the ectopic pregnancy, presence of the foetal heartbeat, presence of free fluid and level of βHCG [5]. Given the limitations of ultrasonography diagnosis of bilateral ectopic pregnancies and lack of formal management guidelines, Jena proposed a management algorithm (Fig. 4) [17].

Fig. 4.

Proposed management algorithm for bilateral tubal pregnancies, adapted from Jena et al. [17].

Surgical options include salpingectomy, salpingostomy, and fallopian tube milking [5]. Salpingectomy is the most common approach to the surgical management of ectopic pregnancy, with the lowest incidence of recurrent ectopic pregnancy for unilateral pathology [5], [18]. However, a bilateral salpingectomy results in sterilisation of the patient. In rural settings, consideration must also be given to the cost and availability of IVF, as well as the surgical expertise available, which may influence the choice of procedure. Studies in 2013 and 2022 reported cases of spontaneous intrauterine pregnancy after unilateral salpingectomy and contralateral salpingostomy; however, there is limited evidence to provide statistics to patients [17], [19], [20].

4. Conclusion

In conclusion, if ectopic pregnancy is suspected, a thorough review of ultrasound images for evidence of bilateral pathology, including multiple corpus luteums, is recommended. Furthermore, given the current limitations in diagnosis of bilateral ectopic pregnancies considered in this report, perioperative discussion regarding possible sterilisation and future fertility wishes is paramount. This is particularly important in rural settings, where clinicians may have less exposure to rare pathologies and more limited access to specialist imaging or fertility services, underscoring the need for heightened vigilance and careful preoperative planning.

Contributors

Eleanor Horsburgh contributed to conception of the case report, acquiring and interpreting the data, and drafting the manuscript.

Penelope Black contributed to patient care, acquiring the data, and revising the article critically for important intellectual content.

Both authors approved the final submitted manuscript.

Patient consent

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

Provenance and peer review

This article was not commissioned and was peer reviewed.

Funding

No funding from an external source supported the publication of this case report.

Declaration of competing interest

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