Triplet heterotopic pregnancy after gonadotropin stimulation and intrauterine insemination diagnosed at laparoscopy: a case report.

Abstract: Heterotopic pregnancy has been traditionally considered a rare event; however, with the use of assisted reproductive technology, the incidence of heterotopic pregnancies is increasing. Diagnosing a heterotopic pregnancy can be challenging. This report describes a 30-year-old female at 11 weeks' gestation with an intrauterine twin pregnancy after gonadotropin stimulation and intrauterine insemination who presented complaining of left lower quadrant abdominal pain with constipation and cramps. The patient was empirically treated for diverticulitis but failed to respond to therapy. A heterotopic triplet pregnancy was ultimately diagnosed at laparoscopy. Heterotopic pregnancy must be considered in the differential diagnosis of abdominal pain in the first trimester, especially in patients who conceived by means of assisted reproductive technology. Surgical treatment is the most frequently used method of treatment.

Key Words: gonadotropin ovarian stimulation, heterotopic pregnancy, multifetal pregnancy

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Heterotopic pregnancy is a potentially fatal condition in which gestation occurs at two or more implantation sites: an intrauterine site and one or more extrauterine sites. It was first described in 1708 by Duverney, (1) who reported the finding at an autopsy of a patient who died as a result of a ruptured ectopic pregnancy but also had a simultaneous intrauterine pregnancy. Heterotopic pregnancy has been reported to occur spontaneously in 1 in 30,000 pregnancies; however, with the use of assisted reproductive technology, the rate may be as high as 1 in 100 pregnancies. (2,3)

Several case reports have described intrauterine twin pregnancies with concurrent heterotopic triplets or quadruplets in the early part of the first trimester. (4-6) Here, we describe a triplet heterotopic pregnancy with intrauterine twins diagnosed in the late first trimester at laparoscopy after empiric treatment for diverticulitis failed to relieve the patient's symptoms.

Case Report

The patient is a 30-year-old [G.sub.4][P.sub.2012] who initially presented for obstetric care at 7 weeks' gestation with an intrauterine twin pregnancy after gonadotropin ovarian stimulation and intrauterine insemination. Early ultrasound examination confirmed the presence of two viable embryos in the uterus; however, the patient had a missed abortion of one of the fetuses at 9 weeks' gestation. At 10 weeks' gestation, the patient presented to an emergency department complaining of left lower quadrant pain, vomiting, and constipation. Urinalysis showed 3+ blood; therefore, a renal ultrasound was performed that showed no evidence of obstruction or nephrolithiasis. Concurrently, the left adnexa were visualized in this study and were reported as normal without evidence of mass. The patient was discharged from the emergency department with stool softeners.

The patient then returned a week later for outpatient follow-up and was found to have persistence of her symptoms. She was hospitalized and a gastroenterology consult was obtained. She was started on intravenous ampicillin-sulbactam, stool softeners, and famotidine for a presumptive diagnosis of diverticulitis. After 48 hours of empiric therapy, the patient had persistence of her symptoms, and a flexible sigmoidoscopy was performed that showed no evidence of colitis or diverticulum. A computed tomography scan of the abdomen and pelvis was then ordered and revealed a 7.5 X 5 cm soft tissue mass in the region of the left adnexa that compressed the sigmoid colon. Transvaginal sonography demonstrated a 5.3 X 7.6 X 10.5 cm solid mass in the left adnexa with the differential diagnosis of an ovarian mass or a pedunculated fibroid (Fig. 1). Because of the size and solid nature of the mass and the symptomatology of the patient, the decision was made to proceed with diagnostic laparoscopic surgery.

[FIGURE 1 OMITTED]

At the time of surgery, a significant hemoperitoneum of 250 mL was found. After clearing the clots from the pelvis, a mass within the left fallopian tube was identified, consistent with an ectopic pregnancy (Fig. 2). The mass was approximately 9.5 X 3 cm and was ruptured approximately 2.5 cm from the cornual region of the uterus. A left salpingectomy was performed through laparoscopy (Fig. 3). The patient recovered well and was discharged home on postoperative day 1 and is still pregnant with the single intrauterine fetus. Histologic examination of the left fallopian tube and its contents revealed chorionic villi, confirming the diagnosis of a triplet heterotopic pregnancy.

Discussion

Heterotopic pregnancies are rare complications of the first trimester; however, the diagnosis is imperative so that treatment can be initiated in a timely manner. A recent case series demonstrated that patients with heterotopic pregnancies are more likely to have tubal rupture and present in hypovolemic shock. (7) In our patient, the diagnosis was delayed because of a confusing clinical presentation. The patient was not acutely ill despite a significant hemoperitoneum from tubal rupture and had primarily bowel complaints with constipation, possibly caused by compression of the sigmoid by the ectopic pregnancy in the left fallopian tube as demonstrated on computed tomography scan. Ectopic and heterotopic pregnancies are frequently diagnosed in the early to middle portion of the first trimester; this case was impressive in that the diagnosis was not made until nearly the second trimester. Also early radiographic studies did not suggest a heterotopic pregnancy.

[FIGURE 2 OMITTED]

[FIGURE 3 OMITTED]

Since heterotopic pregnancy is a rare event, no standardized management recommendations currently exist. Our patient was treated with a laparoscopic salpingectomy; however, other treatment options have been described. Several case reports and series have described successful treatment of a heterotopic pregnancy with transvaginal embryo aspiration of the ectopic under ultrasound guidance followed by local instillation of methotrexate or potassium chloride. (8-10) Surgical treatment remains the most common therapy: laparotomy is frequently used when a patient is hemodynamically unstable and a large hemoperitoneum is suspected; however, laparoscopy can still be successfully and safely used in such situations, as well. (4-7)

Conclusion

In summary, heterotopic pregnancies are becoming more common as the result of increasing numbers of assisted reproductive technology procedures. Early diagnosis is imperative but can be challenging. Surgical treatment with either laparoscopy or laparotomy remains the most common method of management, but nonsurgical modalities have been described.

Acknowledgments

The authors express their gratitude to Karen Roeder for assistance in preparing the figures.

References

1. Duverney JG, in Jombert CA (ed): Oeuvres Anatomiques. Paris, 1708, p 355.

2. DeVoe RW, Pratt JH. Simultaneous intra- and extrauterine pregnancy. Am J Obstet Gynecol 1948;15:1191-1193.

3. Dor J, Seidman DS, Levran D, et al. The incidence of combined intrauterine and extrauterine pregnancy after in vitro fertilization and embryo transfer. Fertil Steril 1991;55:833-834.

4. Chang Y, Lee JN, Yang Ch, et al. An unexpected quadruplet heterotopic pregnancy after bilateral salpingectomy and replacement of three embryos. Fertil Steril 2003;80: 218-220.

5. Nair P, Chauhan M. Heterotopic pregnancy (triplets) following in vitro fertilization: a case report. Arch Gynecol Obstet 1999;263:84-85.

6. Hoopman M, Wilhelm L, Possover M, et al. Case report: heterotopic triplet pregnancy with bilateral tubal and intrauterine pregnancy after IVF. Reprod Biomed Online 2003;6:345-348.

7. Soriano D, Shrim A, Seidman DS, et al. Diagnosis and treatment of heterotopic pregnancy compared with ectopic pregnancy. J Am Assoc Gynecol Laparosc 2002;9:353-358.

8. Oyawoye S, Chander B, Pavlovic B, et al. Heterotopic pregnancy: successful management with aspiration of corneal/interstitial gestational sac and instillation of small dose of methotrexate. Fetal Diagn Ther 2003;18:1-4.

9. Wright A, Kowalczyk CL, Quintero R, et al. Selective embryo reduction in a heterotopic pregnancy using potassium chloride injection resulting in a hematosalpinx. Fertil Steril 1996;66:1028-1030.

10. Porreco R, Burke MS, Parker DW. Selective embryocide in the nonsurgical management of combined intrauterine-extrauterine pregnancy. Obstet Gynecol 1990;75:498-501.

Alex J. Childs, MD, Anthony B. Royek, MD, T. Brian Leigh, MD, and Pamela G. Gallup, MD

From the Department of Obstetrics and Gynecology, Mercer University School of Medicine, Memorial Health University Medical Center, Savannah, GA.

Presented as a poster presentation at the Georgia and South Carolina OB/GYN Society Annual Meeting at Hilton Head, SC, September 9 to 12, 2004.

No financial support was received for this article.

The authors do not have any commercial or proprietary interest in any drug, device, or equipment mentioned in this article.

Reprint requests to Dr. Alex J. Childs, Department of Obstetrics and Gynecology, 4700 Waters Ave., Savannah, GA 31404. Email: [e-mail]

Accepted February 14, 2005.

RELATED ARTICLE: Key Points

* Heterotopic pregnancy is a rare occurrence; however, with the use of assisted reproductive technology, the incidence is increasing.

* The diagnosis of heterotopic pregnancy must be considered in first-trimester pregnant patients who conceived with assisted reproductive technology and present with abdominal pain.

* Several treatment modalities have been described for heterotopic pregnancies, but surgical treatment remains the most common.