Ruptured heterotopic pregnancy with successful term intrauterine pregnancy outcome after natural conception: a case report

Abstract

Background

Heterotopic pregnancy is the simultaneous existence of intrauterine and extrauterine pregnancies. This is a rare obstetric case, with an incidence of 1 in 30,000 in naturally conceived pregnancies and 1 in 100 pregnancies achieved through in vitro fertilization. Early diagnosis of heterotopic pregnancy is challenging in developing countries with no high-resolution ultrasound.

Case presentation

A 22-year-old multigravida Somali mother presented to our hospital on her tenth week of gestation with the complaint of progressively worsening abdominal pain of 7 days’ duration. The patient had type III female genital mutilation at the age of 7 years. The patient presented with tachycardia, but her overall hemodynamic status remained stable. Obstetric ultrasound showed a right-sided adnexal mass, a hemoperitoneum, and an intrauterine pregnancy at 10 weeks + 2 days of gestation. Two units of screened, cross-matched whole blood were prepared, and the patient underwent an exploratory laparotomy. Intraoperative findings revealed a right-sided ruptured tubal ectopic pregnancy at the ampulla with active bleeding, a hemoperitoneum of approximately 900 mL with clots, and a bulky uterus about 10 weeks in size with a normal left tube and ovary. The patient had a brisk recovery postoperatively, and the intrauterine pregnancy was conservatively monitored until term gestation. An alive female neonate weighing 3000 gm was delivered via spontaneous vertex delivery with an Apgar score of 8 and 9 in the first and fifth minutes, respectively.

Conclusion

Heterotopic pregnancy is a rare obstetric condition that can occur through natural conception. Female genital mutilation might be an independent risk factor for heterotopic pregnancy that occurs via natural conception. The presence of an intrauterine gestation does not rule out a concomitant extrauterine gestation, so a high index of suspicion and timely diagnosis should be made for a pregnant woman presenting with acute abdominal pain and an adnexal mass. A routine early-first-trimester antenatal ultrasound scan of the adnexa by an experienced obstetrician or sonographer is paramount to obtaining an accurate and timely diagnosis.

Introduction

Heterotopic pregnancy (HP) is a rare obstetric condition in which at least two pregnancies occur concurrently at different implantation sites, with a prevalence of 1 in 30,000 through natural conception to around 1 in 100 through assisted or in vitro reproduction. It typically manifests as simultaneous intrauterine (IUP) and extrauterine (ectopic) pregnancies [1–6]. The most common ectopic site is the fallopian tube, both in spontaneous and assisted heterotopic pregnancies. The cornual site is the second most common site, while the cervix, ovary, and abdominal HP are extremely rare [7–9].

The clinical presentations and risk factors of HP mimic those of ectopic pregnancy. The predisposing factors include early age of sexual intercourse, increasing maternal age, multiple sexual partners, pelvic infections, history of infertility and use of fertility drugs, previous ectopic pregnancies, and previous pelvic surgeries such as female genital mutilation (FGM) [7–10]. The rate of detection of HP decreases from 70% at around 5–8 weeks of gestation to about 10% after the 11th week of gestation [7–9]. However, the presence of a viable IUP during ultrasonographic scanning can mislead the physician into detecting the extrauterine component of HP [4, 11].

Despite the continued increase in the incidence of ectopic pregnancy, the rate of death from ectopic pregnancy has declined in developed countries primarily due to earlier diagnosis before tubal rupture, which occurs in over 70% of cases. On the contrary, in developing countries, the diagnosis of HP is difficult and usually delayed, presenting as a ruptured ectopic pregnancy. The misdiagnosis of heterotopic gestation is more common in developing countries due to the low resolution of the ultrasound to identify an early ectopic gestation [4, 11–16].

The delayed diagnosis can result in increased rates of morbidity and mortality, both for the mother and intrauterine gestation [3, 4, 12, 17, 18]. In almost 70% of heterotopic pregnancies, the intrauterine conceptus has a good outcome similar to that of singleton pregnancies [3].

We, therefore, report a case of ruptured heterotopic pregnancy presenting at the 10th week of gestation following delayed diagnosis and term vertex delivery after exploratory laparotomy and salpingectomy of ruptured tubal ectopic pregnancy was done.

Case presentation

A 22-year-old Somali mother (gravida 2 para 1), in her tenth week of gestation, presented with lower abdominal pain and vomiting of 1 weeks’ duration that became severe over the last 2 days. The abdominal pain was cramping in nature and located in the suprapubic area, but subsequently increased in intensity and became generalized. The patient had a history of FGM at the age of 7 years. She has no personal or family history of congenital anomalous conditions or medical conditions like diabetes mellitus, hypertension, cardiac illness, renal disease, or similar clinical conditions. The patient did not have any history of pelvic infections or sexually transmitted diseases (STDs).

On physical examination, she was fully conscious and in pain; her blood pressure was 100/60 mmHg with a pulse rate of 110 beats/minute, and her respiratory rate was 24 cycles/minute. She had a slightly pale conjunctiva. There was generalized abdominal tenderness and guarding with suprapubic fullness. The vulva and vagina were clear with no bleeding, the cervix was closed, and the pouch of Douglas was full. She also had positive cervical motion tenderness.

Emergency obstetric ultrasound showed a viable intrauterine pregnancy with a right adnexal echo-complex mass measuring 6 × 6 cm and free fluid in the cul-de-sac. She was then admitted to the gynecology emergency ward with the diagnosis of first-trimester pregnancy plus a ruptured hemorrhagic ovarian cyst.

The consultant physician re-evaluated the patient, and the ultrasound done yielded a viable intrauterine embryo with a crown‑rump length of 10 weeks plus 2 days, a right adnexal echo-complex mass with increased blood flow and free fluid in the posterior cul-de sac, pericolic gutters, and Morrison’s pouch; the diagnosis of ruptured ectopic with viable intrauterine gestation (i.e., heterotopic gestation) was made (Fig. 1). Laboratory results showed an initial hematocrit of 32.6%, which later became 23.6%.

Fig. 1.

Ultrasonographic images showing (A) a viable intrauterine pregnancy, free fluid in the posterior cul-de sac, pericolic gutter, and Morrison’s pouch (indicated with arrows) and (B) a right adnexal echo-complex mass with increased blood flow (arrow)

Two units of screened, cross-matched whole blood were prepared, and the patient was counseled for an exploratory laparotomy. After written informed consent was obtained, an exploratory laparotomy was done under general anesthesia. Intraoperative findings revealed a right-sided ruptured tubal ectopic pregnancy at the ampulla with active bleeding (Fig. 2), a hemoperitoneum of approximately 900 mL with clots, and a bulky uterus about 10 weeks in size with a normal left tube and left ovary. The total estimated blood loss was 1000 mL. Right-sided salpingectomy was done, and hemostasis was secured.

Fig. 2.

A right-sided ruptured tubal ectopic pregnancy at the ampulla with active bleeding (arrow)

The excised mass was sent for histopathology analysis. Gross examination showed a 6 cm-long salpingectomy specimen that was friable in consistency, with a 4 cm × 3 cm × 3 cm opened-up clot-filled proximal segment (Fig. 3). The microscopic section showed fallopian tube tissue with variable sizes of chorionic villi, hemorrhage, necrosis, and product of conception (Fig. 4). The pathologic diagnosis confirmed a right fallopian tube ectopic pregnancy.

Fig. 3.

Gross salpingectomy specimen showing a 6 cm-long speciman, friable in consistency, with 4 cm × 3 cm × 3 cm opened-up clot-filled proximal segment

Fig. 4.

Microscopic sections of the excised specimen. (A and B) High-powered magnification showing chorionic villi with central fibrovascular stroma and outer trophoblastic cell layer. (C) A low-powered view showing chorionic villi with surrounding fibrin and hemorrhage

The patient tolerated the surgery and had a brisk recovery. The intrauterine pregnancy remained uneventful, and she was on regular antenatal visits until term gestational age. She delivered an alive female neonate weighing 3000 gm via spontaneous vaginal delivery with an Apgar score (denoting appearance, pulse, grimace, activity, and respiration) of 8 and 9 in the first and fifth minutes, respectively. Exclusive breastfeeding was immediately initiated. She was then counseled on the need for early antenatal booking and confirming pregnancy as early as the fifth week of gestation in the next pregnancy, as there is an increased risk of recurrence of ectopic pregnancy. The mother was then discharged and linked to a family planning clinic after adequate counseling was given, and the baby was linked to an infant and child clinic for an immunization schedule.

Discussion

Heterotopic pregnancy is an extremely rare phenomenon through natural conception [12]. However, it tends to be more prevalent in pregnancies achieved through assisted reproduction, reaching as high as 5% following in vitro fertilization [1–6]. In our case, the patient conceived naturally. In this case, the fallopian tube was the ectopic implantation site, which is consistent with most HP cases [7–9]. The rate of HP detection decreases as the gestational age goes by since the growing uterus will obscure the detection of the extrauterine component [7–9]. In this case, the patient presented at the tenth week of gestation.

In this case, there was no identified major risk factor apart from FGM, a rare factor that could potentially predispose the patient to repetitive pelvic infection and/or ectopic pregnancy [7–10]. HP tends to be symptomatic in the first trimester of pregnancy, usually from 5 to 10 weeks of gestation, and the clinical presentations mimic those of ectopic pregnancy [7–9]. However, it can occasionally remain asymptomatic in the second and third trimesters of HP. Unfortunately, by the time the patient becomes symptomatic, the rupture has occurred. In this case, the patient presented with lower abdominal pain, vomiting, generalized abdominal tenderness, guarding, and suprapubic fullness with no sign of hypovolemic shock.

The greatest challenge of heterotopic pregnancy is making a diagnosis. While diagnostic laparoscopy with direct visualization remains the gold standard, transvaginal Doppler ultrasound is the preferred noninvasive alternative, with a sensitivity and specificity of 84% and 98%, respectively. Using transabdominal ultrasound will raise the sensitivity for the diagnosis of an ectopic pregnancy from 53% to 73%. A transabdominal Doppler ultrasound will show increased blood flow characterized by a high-velocity fire pattern in the adnexal mass [4, 11–16]. In this case, the diagnosis was made via transabdominal ultrasound, and it showed increased blood flow (Fig. 1).

Moreover, prompt surgical intervention should be sought in a woman with an acute abdomen and hemoperitoneum, even in the face of diagnostic uncertainty. Conservative surgical management allows the viable intrauterine pregnancy to develop to term, ultimately leading to delivery [4]. This case demonstrated that laparotomy can be a successful treatment modality for the extrauterine part of heterotopic pregnancy with conservative follow-up to the intrauterine conceptus (Fig. 2).

The timing of the diagnosis of HP is vital for the prognosis of both the mother and fetus, as delay in diagnosis or misdiagnosis can result in an increased risk of morbidity and mortality for the mother and intrauterine gestation. In our case, the diagnosis was made late, as she presented with a ruptured ectopic pregnancy. In almost 70% of the cases, the intrauterine conceptus of heterotopic pregnancies has a good outcome similar to that of singleton pregnancies when the diagnosis is made before the rupture of the ectopic pregnancy [3, 13]. In our case, the outcome was an alive female neonate weighing 3000 gm with an Apgar score of 8 and 9 in the first and fifth minutes, respectively.

Conclusion

Heterotopic pregnancy remains a rare obstetric condition, especially through natural conception. Female genital mutilation might be an independent risk factor for heterotopic pregnancy that occurs via natural conception. However, a high index of suspicion and timely diagnosis should be made for a pregnant woman who presents with acute abdominal pain and an adnexal mass, even with no identifiable risk factors. A routine early first-trimester antenatal ultrasound scan of the adnexa by an experienced obstetrician or sonographer is paramount to obtain an accurate diagnosis and proper management. Awareness of preconception health and future fertility is very important, particularly in a resource-limited country. Moreover, conservative surgical management allows the viable intrauterine pregnancy to develop to term, ultimately leading to a good intrauterine pregnancy outcome.

Abbreviations

HP

Heterotopic pregnancy

FGM

Female genital mutilation

IUP

Intrauterine pregnancy

Author contributions

All authors made a significant contribution to the work reported, whether that was in the conception, study design, execution, acquisition of data, analysis, and interpretation, or all these areas. They also took part in drafting, revising, or critically reviewing the article and/or gave final approval of the version to be published. They have agreed on the journal to which the article will be submitted and agreed to be accountable for all aspects of the work.

Funding

The authors did not receive any funding for this case report.

Data availability

All datasets generated and/or analyzed during the current study are included in this case report.

Declarations

Ethics approval and consent to participate

Not applicable. Our institution does not require approval for the publication of case reports.

Consent for publication

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Competing interests

The authors declare no conflicts of interest regarding the publication of this study.