Laparoscopic Surgery for Heterotopic Pregnancies: Two Cases

Introduction

Heterotopic pregnancy is defined as the simultaneous occurrence of both an intrauterine and an extrauterine pregnancy. The frequency of heterotopic pregnancy is reported as 1:7963–1:30,000. ¹ The incidence of heterotopic pregnancy after ovulation induction medication was reported to be 33:10,000, but its incidence after assisted reproductive techniques (ART) is as high as 100:10,000, and is likely to increase with the spread of ART procedures.^2,3 Two successfully treated cases of ruptured heterotopic pregnancies, one after intrauterine insemination (IUI) and the other after in vitro fertilization (IVF), are reported here. These cases illustrate that the presence of an intrauterine pregnancy does not rule out the possibility of ectopic pregnancy. In addition, laparoscopy can be considered a potent alternative for ruptured ectopic pregnancy with hemoperitoneum.

Cases

Case 1

A 31-year-old woman, gravida 0, presented to the department of obstetrics and gynecology, in the Research Institute of Clinical Medicine, at Chonbuk National University Medical School, complaining of infertility of 1 year's duration. The patient underwent intracytoplasmic sperm injection (ICSI) and embryo transfer (ET) for male factor infertility. Eighteen days after ET, at 4 and 3/7 weeks of gestation, the patient complained of mild abdominal discomfort and slight vaginal bleeding. Ultrasonography showed no intrauterine gestational sac, with enlarged multicystic ovaries and moderate ascites. Her serum concentration of β-human chorionic gonadotropin (hCG) was 834.0 international units (IU)/mL. Twenty-seven days after ET, at 5 and 3/7 weeks of gestation, the patient complained of severe abdominal pain and vaginal bleeding. At that time, her β-hCG level reached 4126.5 IU/mL. Ultrasonography revealed increased free fluid in the abdomen. Diagnostic laparoscopy for the differential diagnosis of hemoperitoneum was recommended. A large quantity of hemoperitoneum and a ruptured left ampullar ectopic pregnancy were found (Fig. 1). A left salpingectomy was performed. The pathology report of the resected specimen confirmed the presence of chorionic villi. The patient's postoperative period was uneventful. At 39 and 1/7 weeks of gestation, the patient delivered a healthy newborn weighing 3910 g.

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FIG. 1.

Laparoscopic view of an ectopic tubal pregnancy (arrow) with bilateral hyperstimulated ovaries.

Case 2

A 32-year-old woman, gravida 2, para 0, presented with complaint of severe abdominal pain of 6 hours' duration, and mild vaginal bleeding at 7 and 3/7 weeks after her last menstrual period. The patient had a history of a first trimester surgical termination of pregnancy and a unilateral left salpingectomy for a spontaneous ectopic pregnancy. She underwent IUI at another medical center. On admission, she was pale, with a blood pressure of 100/60 mm Hg and a pulse rate of 120. Abdominal examination revealed abdominal distension and diffuse abdominal tenderness with signs of peritoneal irritation. Transvaginal sonography revealed a single intrauterine gestational sac and an abnormal echo complex mass in the left adnexa (Fig. 2A and B). In addition, a moderate amount of fluid was seen in the cul-de-sac. Her serum β-hCG level was ≥100,000 milli-IU (mIU)/mL.

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FIG. 2.

Pelvic ultrasound. (A) A viable intrauterine pregnancy is noted. (B) An abnormal complex mass in the left adnexa is noted (arrow).

An emergency laparoscopy was performed with the diagnosis of heterotopic pregnancy. The laparoscopy revealed ∼500 mL of hemoperitoneum and a ruptured left ampullar pregnancy, hence left salpingectomy was performed. The histopathologic results confirmed the diagnosis of a left tubal pregnancy. The patient had an uneventful postoperative recovery and was discharged on the 6th postoperative day. At 9 and 4/7 weeks of gestation, an ultrasound scan confirmed a viable intrauterine pregnancy. Since then, the patient has been lost to follow-up.

Results

The first patient, at 39 and 1/7 weeks of gestation, was delivered of healthy newborn, weighing 3910 g. In the second patient, at 9 and 4/7 weeks of gestation, an ultrasound scan confirmed a viable intrauterine pregnancy. Since then, the patient has been lost to follow-up.

Discussion

Heterotopic pregnancy, in which there are simultaneous intrauterine and extrauterine gestations, is a rare condition, but with the widespread use of ART, it will become increasingly more common.

The preoperative diagnosis of heterotopic pregnancy can be a major challenge to clinicians because of its rarity and the wide variability of its presentations. Marcus et al. reported the clinical presentations of the 20 cases of heterotopic pregnancies in which 45% of the patients were asymptomatic; 30% of them complained of abdominal pain and vaginal bleeding, and 25% had no vaginal bleeding. ⁴

Serial concentrations of serum β-hCG may also be helpful in the diagnosis of ectopic pregnancy, but these values are often unreliable with heterotopic pregnancy because subnormal hormone production from ectopic pregnancy may be masked by the higher placental production from an intrauterine pregnancy. ⁵ Transvaginal ultrasound can be an important aid. However, sonographic identification of an ectopic pregnancy has low sensitivity (0.56). ⁶ The diagnosis of a heterotopic pregnancy by ultrasound findings alone, in the absence of any clinical abnormality was made in 54% of cases in a literature review. ⁷ According to Tal et al., only 41.1% of cases could be diagnosed with the combination of clinical and ultrasound findings. ⁸ During an ultrasound examination, heterotopic pregnancy is initially misdiagnosed, with a corpus luteal cyst assumed to be an etiology for intraperitoneal fluid and clinical deterioration rather than an ectopic pregnancy, especially if the concurrent intrauterine pregnancy is proceeding well. ⁹ Furthermore, among patients undergoing IVF, the presence of severe ovarian hyperstimulation syndrome (OHSS) may obscure the diagnosis of hemoperitoneum-related conditions, such as an ectopic pregnancy or a ruptured corpus luteal cyst, therefore leading to a delayed diagnosis. In the first case, the presence of free fluid in the pelvis was initially mistaken for ascites, which are a common finding in OHSS, and the heterotopic pregnancy was then diagnosed after rupture of the ectopic pregnancy. Even with a high index of suspicion, most heterotopic pregnancies are diagnosed after rupture of the ectopic pregnancy. ¹⁰ Because transvaginal ultrasound may not be a reliable tool for diagnosing an ectopic pregnancy with concomitant OHSS, a more effective diagnostic tool to improve the diagnosis is required. ¹⁰

The difficulty of an accurate clinical and sonographic diagnosis in symptomatic patients can justify laparoscopy.^7,10 The conventional treatment for heterotopic pregnancy is laparoscopy or laparotomy with minimal manipulation of the uterus. Laparoscopy may be an appropriate method for managing patients with heterotopic pregnancies to maintain the intrauterine pregnancies. ⁷ Laparotomy is reserved for cases with serious intraperitoneal bleeding or hemodynamically instability caused by hemorrhagic shock.^7,11 However, in the cases described here, the heterotopic pregnancies were easily removed laparoscopically after evacuation of blood from the abdominal cavity. Odejinmi et al. reported that operative laparoscopy is safe and sustainable in most women with ectopic pregnancies with hemodynamic instability, but the procedure requires substantial laparoscopic skill. ¹² With respect to the prognosis of intrauterine pregnancy, favorable outcomes are reported in 66% of cases of heterotopic pregnancies treated mainly by surgery. ⁸ In the last couple of decades, conservative nonsurgical treatment such as local injection of potassium chloride or methotrexate has been selected as an alternative treatment for unruptured heterotopic pregnancy. ¹³ However, the risks of potential adverse effects on the intrauterine gestation and rupture of the ectopic pregnancy should be kept in mind with nonsurgical management. Table 1 summarizes the cases of heterotopic pregnancies reported in the literature, including the present cases.

Conclusions

The presence of an intrauterine pregnancy and OHSS may result in a delay in diagnosis of a heterotopic pregnancy, as in the case reported here. This case report highlights that clinicians need early clinical and careful sonographic assessment of the whole pelvis, especially in patients undergoing IVF, as this measure may permit earlier diagnosis before rupture. Furthermore, laparoscopy in the presence of hemoperitoneum but hemodynamic stability is a good diagnostic tool for uncertain pregnant conditions, and a prudent minimally invasive procedure to preserve the developing intrauterine pregnancy.