Dual extrauterine ectopic pregnancy: double management

Abstract

A 30-year-old nulliparous woman was referred with suspected left ovarian ectopic pregnancy. She had undergone laparoscopic left salpingectomy for ruptured tubal ectopic pregnancy 3 weeks earlier, following treatment with medications for ovulation induction. Sonological examination revealed a left ovarian ectopic pregnancy corresponding to 8 0/7 weeks with cardiac activity. She underwent ultrasound-guided intrasac therapy with intrasac instillation of 3 mEq of potassium chloride followed by 50 mg of methotrexate. She was followed with weekly measurements of serum beta human Chorionic Gonadotropin (hCG) which returned to baseline after 65 days of the intrasac therapy. This case not only highlights the need for continued follow-up of the serum beta hCG after definitive management of an ectopic pregnancy in cases with multiple ovulations, but also the option of medical management in cases of advanced ovarian ectopic pregnancy. It also accentuates the necessity for adequate counselling to avoid conception in a multiple ovulation cycle.

Background

The occurrence of multiple concurrent ectopic pregnancies is very rare, the incidence of which is unknown. Multifollicular development accompanies with it the risk of multiple ovulations and thence multiple ectopic pregnancies. It is indeed a diagnostic challenge to detect concomitant ectopic pregnancies specifically in a situation of a concurrent tubal and ovarian ectopic pregnancy. The diagnosis is arduous because of the sonological similarity between an ovarian ectopic pregnancy and corpus luteum in the absence of fetal pole or yolk sac. The case illustrated here highlights the rare risks of multiple ectopic pregnancies associated with multiple ovulations. Understanding this possibility is prudent to prevent life-threatening consequences including maternal morbidity and mortality.

Case presentation

A 30-year-old nullipara in a monogamous relationship for 1 year was referred with a suspected left ovarian ectopic pregnancy. Her history was significant for laparoscopic left salpingectomy, which was performed 3 weeks earlier for ruptured tubal ectopic pregnancy. She presented with pain in the left abdomen and vaginal bleeding of 1 day duration. On clinical examination, she was haemodynamically stable with no demonstrable tenderness or guarding of the abdomen. The speculum examination showed evidence of blood at the external os, and bimanual examination revealed a normal uterus with left adnexal fullness and tenderness.

The patient’s medical history was significant for infertility with intake of medications for ovulation induction with aromatase inhibitors and gonadotropin injection’s 8 weeks prior to her presentation. This had resulted in the development of five dominant follicles and was allowed for a natural relation cycle. Five weeks later, she presented with symptoms of progressively increasing pain in the left iliac fossa associated with vaginal bleeding of 2 days. Sonogram findings were significant for a gestational sac in the left adnexa measuring 22×19×20 mm³ and free fluid in the posterior cul-de-sac. Laboratory values were remarkable for serum beta hCG level of 6539 mIU/mL. A diagnosis of ruptured left tubal ectopic pregnancy was made and she underwent an emergency laparoscopic left salpingectomy. Per operatively a left isthmic ruptured ectopic pregnancy with normal right fallopian tube and bilateral ovaries was noted and the histopathology had confirmed the left tubal pregnancy.

Investigations

Laboratory values were significant for a serum beta hCG of 44,179 mIU/mL. The remaining biochemical and haematological parameters were within the normal limits. Transvaginal ultrasound illustrated a normal empty uterus with a well-defined gestational sac measuring 28×25×22 mm³ in the left ovary and an adjacent corpus luteum. There was no ambiguity in the diagnosis as a fetal pole with crown-rump length of 15.5 mm corresponding to 8 0/7 weeks with cardiac activity was clearly visualised in the intra ovarian gestational sac. The power doppler revealed a vascular ring around the ectopic sac analogous to the adjoining corpus luteum. The right ovary appeared normal and there was no evidence of fluid in the posterior cul-de-sac (figure 1).

Figure 1.

This figure illustrates left ovarian ectopic pregnancy with fetal pole and cardiac activity with a corpus luteum adjacent to it.

Differential diagnosis

In view of her antecedent history of ruptured tubal ectopic pregnancy and the acute nature of her symptoms, the initial workup was aimed at detecting a second ectopic pregnancy and ruling out ovarian torsion and tubo-ovarian mass. A missed concurrent ectopic pregnancy was more likely than the differentials’ of ovarian torsion and tubo-ovarian mass considering her history of ovulation induction. The transvaginal ultrasound findings corroborated the diagnosis of an ovarian ectopic pregnancy.

Treatment

The patient was counselled on the diagnosis and management options. She was planned for conservative management although it was an advanced ovarian ectopic pregnancy. This was essentially to conserve her ovary in view of her young age and nulliparity status, concurrently avoiding a second surgery. She also consented for emergency surgery in the possibility of an unsuccessful medical management. Transvaginal ultrasound guided intrasac therapy was performed with a 17 gauge, 35 cm long, single lumen ovum aspiration needle. Disappearance of cardiac activity was confirmed after intrasac instillation of 3 mEq of potassium chloride. The gestational sac contents were aspirated using a 20 cm³ syringe followed by instillation of 50 mg methotrexate into the sac (figure 2).

Figure 2.

This figure illustrates the intrasac therapy with potassium chloride and methotrexate in the management of ovarian ectopic pregnancy using ovum aspiration needle.

Outcome and follow-up

On follow-up, her serum beta hCG dropped to 13,558 mIU/mL after 1 week and a value of 3 mIU/mL was noted after 65 days of the intrasac therapy. The patient recovered uneventfully and on follow-up after 3 months, a normal antral follicular count was noted which confirmed that this conservative approach had not affected her ovarian reserve.

Discussion

A concurrent tubal and ovarian ectopic pregnancy following ovulation induction is very rare, the incidence of which is unknown. A vast majority of the ectopic pregnancies occur in the fallopian tube with the ovarian site being one of the rarest presentations accounting for 0.3%–3% of all ectopic pregnancies. The ovarian ectopic pregnancy is sonologically similar to corpus luteum which makes its detection challenging, often leading to misdiagnosis in majority of the cases.^{1 2} Multifollicular development and consequent multiple ovulations with ovulation induction medications increases the risk of multiple pregnancies, but there are very few case reports of multiple concomitant ectopic pregnancies in the literature.

During the woman’s initial presentation, the diagnosis of ovarian ectopic pregnancy was blurred because of the presence of a concomitant tubal ectopic pregnancy. The clinical signs and symptoms of an ovarian ectopic pregnancy are similar to that of a tubal pregnancy, and the diagnosis is made after histopathological examination as the intra operative diagnosis of ovarian pregnancy is possible in only 28% of the cases.³ A century ago Otto Spiegelberg described four main criteria that remain the standard in diagnosing a primary ovarian pregnancy to this day: (1) an intact ipsilateral tube, clearly separate from the ovary; (2) a gestational sac occupying the position of the ovary; (3) a gestational sac connected to the uterus by the ovarian ligament; and (4) ovarian tissue in the wall of the gestational sac. Comstock et al has described the sonographic characteristics of ovarian pregnancies as thick-walled, echogenic rings with anechoic centre that are located within or on the surface of the ovary. This differentiates them from a tubal ectopic ring, which is much thinner by comparison. The increased echogenicity of the ovarian ectopic pregnancy compared with the ovarian stroma differentiates it from a corpus luteal cyst, which is usually less echogenic.⁴ Even with all the technological advances there is still a possibility of missing an ovarian pregnancy because of the great degree of overlap with the more common tubal ectopic pregnancy and corpus luteal cyst. The diagnosis of an ovarian ectopic pregnancy is difficult because an embryo is seen in less than 10% of sonographically detected ovarian ectopic pregnancy.⁵

The occurrence of the concomitant ovarian and tubal pregnancies after ovulation induction is due to multifollicular development. Case reports on dual ectopic pregnancies following double embryo transfer⁶ and following controlled ovarian hyperstimulation and intrauterine insemination cycle⁷ have been reported both of which were managed surgically. Given the relative frequency of tubal ectopic pregnancies in the infertile population, the importance of choosing an appropriate management strategy is further underscored knowing the morbidity and potential mortality of ruptured ectopics within the context of the patient’s reproductive goals. There are established criteria for management of ectopic pregnancies, which include pharmacologic, surgical and expectant management under specific circumstances. However, there are no well-defined studies or data to suggest standard of care in the case of concomitant ectopic pregnancies in different extrauterine sites, particularly in the setting of coexisting ruptured and unruptured ectopic pregnancies. Early diagnosis and treatment are absolutely necessary to ensure a successful outcome. In contrast to tubal ectopic pregnancy, medical treatment by methotrexate has not been a routine option for ovarian ectopic pregnancies.^{8 9} The mainstay of management for ovarian pregnancy is oophorectomy or conservative surgery such as ovarian wedge resection depending on the extent of tissue destruction.^{10 11} Successful medical management of early ovarian ectopic pregnancy has been reported and is an excellent option for conserving the ovary.¹²

This is the first case report with conservative management of an advanced ovarian ectopic pregnancy following surgical management of a concomitant tubal pregnancy. We performed medical management for this advanced ovarian ectopic pregnancy after counselling, chiefly to conserve the ovary and to avoid a second surgery.

Patient’s perspective.

The whole experience was very frightening initially as I had already undergone a surgery with removal of my left fallopian tube. The diagnosis of another ectopic was a nightmare but when I was offered medical management I was relieved mainly because I need not undergo another surgery and also as doctors said they would be conserving my ovary. I was put on an intense follow-up after treatment and now I am happy because i am completely cured of my ectopic pregnancy without undergoing another surgery.

Learning points.

• A high index of suspicion for multiple pregnancies and multiple ectopic pregnancies in ovulation induction cycles particularly with multifollicular development is a must.

• In cases of multifollicular development in ovulation induction cycles, follow-up of the serum bhCG values even after definitive management of an established ectopic pregnancy is important for early detection of rare multiple ectopic pregnancies.

• Fertility units should develop guidelines to ensure single follicular development with ovulation induction medications and promote cycle cancellation with adequate counselling against conception in the event of multifollicular development.

• The option of fertility-sparing medical management should be considered and discussed with the patient before embarking on major surgical intervention in cases of advanced ovarian ectopic pregnancy.

Ethics statements

Patient consent for publication

Consent obtained directly from patient(s).