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Year : 2017  |  Volume : 6  |  Issue : 2  |  Page : 126-129

Heterotopic pregnancy: A report of three cases and a brief comparative review

Department of Obstetrics and Gynecology, Military Training Hospital Mohammed V, Rabat, Morocco

Date of Web Publication15-Sep-2017

Correspondence Address:
Moukit Mounir
Department of Obstetrics and Gynecology, Military Training Hospital Mohammed V, Hay Riyad, 10100, Rabat
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/sjhs.sjhs_58_17

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Heterotopic pregnancy (HP) is a rare event combining intrauterine pregnancy (IUP) and extrauterine pregnancy. It can occur in the absence of any predisposing risk factors. We present three cases of HP managed in our department, two in a natural conception and the other following ovulation induction. The three cases had the same clinical presentation and surgical treatment. Preservation of the IUP was obtained in two cases. Unfortunately, the other case was aborted spontaneously on the 2nd postoperative day. Only with an early diagnosis and adequate management, the IUPs will reach viability with a great chance of a favorable obstetric outcome.

Keywords: Heterotopic pregnancy, prophylactic tocolysis, surgery, transvaginal ultrasound

How to cite this article:
Jaouad K, Mounir M, Rachid A, El Mehdi EM, Abdellah B, Driss MR, Mohammed D. Heterotopic pregnancy: A report of three cases and a brief comparative review. Saudi J Health Sci 2017;6:126-9

How to cite this URL:
Jaouad K, Mounir M, Rachid A, El Mehdi EM, Abdellah B, Driss MR, Mohammed D. Heterotopic pregnancy: A report of three cases and a brief comparative review. Saudi J Health Sci [serial online] 2017 [cited 2021 Jan 28];6:126-9. Available from: https://www.saudijhealthsci.org/text.asp?2017/6/2/126/214852

  Introduction Top

Heterotopic pregnancy (HP) is a rare form of twin pregnancy defined as the simultaneous presence of intrauterine pregnancy (IUP) and extrauterine pregnancy. It can follow an assisted or a natural conception cycle.[1] Significant morbidity and occasional mortality have been reported as a result of a delay in diagnosis. The objective of this article is to raise the awareness of an increase in incidence of spontaneous HP and to discuss the best practices for the diagnosis and treatment of this rare entity.

  Case Reports Top

Case 1

A 32-year-old woman, gravida 2, parity 1, presented to the gynecological emergencies with the chief complaint of lower abdominal pain appeared 48 h before. At this time, the patient was pregnant at 6th week of gestation. She conceived spontaneously, and she had no history of pelvic inflammatory disease (PID). Physical examination showed mild tenderness at the right lower abdomen; pouch of Douglas was painful on palpation with no bleeding from the uterine cervix. Transvaginal ultrasonography objectified simultaneous intrauterine and extrauterine gestational sacs according to 6 weeks of gestation with current cardiac activity of both embryos [Figure 1]. The patient was consented for an operative laparotomy few hours later. A volume of 200 mL of hemoperitoneum was aspirated. The uterus was enlarged according to the gestational age. The left annex and right ovary were normal with a ruptured ectopic pregnancy in the ampullary region of the right fallopian tube. A right salpingectomy was performed and the material was sent for histological examination confirming chorionic villi. Nicardipine (Loxen ®) was used for tocolysis. The IUP proceeded well, and the labor was performed at term with a healthy baby.
Figure 1: Transvaginal ultrasound showing simultaneous intrauterine and ectopic pregnancy at 6 weeks of gestation

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Case 2

A 25-year-old woman was a case of primary infertility for 2 years and ultrasound findings of polycystic ovary syndrome. Hysterosalpingography and her spouse spermogram were normal. She received ovulation induction by clomiphene citrate (CC) 100 mg/day starting on day 2 of the cycle and continued with recombinant follicle-stimulating hormone (FSH) on day 7. Transvaginal ultrasound on day 12 showed two good-sized follicles in the right ovary; the left ovary contained one good-sized follicle in addition to other small follicles <10 mm. Endometrium was 12-mm thick. She was given human chorionic gonadotropin (HCG) on day 12 and was advised for natural intercourse on day 13. Her next presentation was at 6 weeks of amenorrhea with lower abdominal pain. On admission, the patient was afebrile with normal vital signs. On physical examination, mild tenderness was found on deep palpation of the right lower abdominal quadrant. Transvaginal ultrasound scan revealed intrauterine gestational sac with fetal pole and visible cardiac activity. A small amount of free fluid was seen in the pouch of Douglas. Given her history of fertility treatment and her physical examination findings, adnexal areas were evaluated with particular detail. A second gestational sac with fetal pole and cardiac activity, adjacent to the right ovary, was visualized, suggestive of HP [Figure 2]. An urgent minilaparotomy was performed; there was a preruptured ectopic pregnancy in the ampullary region of the right fallopian tube. Partial salpingectomy was performed. Pathological examination confirmed the ectopic pregnancy. Peroperatively, intravenous nicardipine was prescribed for the prevention of uterine contractions. The IUP was followed up till 41 weeks, and she was induced (one dose of misoprostol) with vaginal delivery of a healthy baby male neonate.
Figure 2: Simultaneous intrauterine and ectopic pregnancy

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Case 3

A 21-year-old woman, with a history of right ectopic pregnancy which was treated with methotrexate 2 years before, presented in our department for acute abdominal pain with amenorrhea for 2 months. Urine pregnancy test, made 10 days before, confirmed her pregnancy. On examination, the patient had a blood pressure of 90/60 mmHg, pulse of 110 beats/min, and there was a generalized abdominal distention with tenderness. Gynecological examination revealed closed cervix with vaginal bleeding. Transvaginal ultrasonography showed a middle amount of free fluid in the peritoneal cavity, an intrauterine gestational sac without fetal pole, and a right heterogeneous mass, measuring 25 mm. Her hemoglobin was 8 g/dl. She was planned for urgent minilaparotomy with the differential diagnosis of IUP with either ruptured corpus luteum or hemorrhagic ovarian cyst, thinking least of HP. Laparotomy revealed approximately 500 mL of hemoperitoneum and a ruptured right cornual pregnancy; enucleation of the cornual pregnancy was performed with right salpingectomy [Figure 3]. She received 2 units of blood after surgery raising the postoperative hemoglobin to 10 g/dl. Unfortunately, she aborted spontaneously on the 2nd postoperative day, and she was discharged from the hospital on the 5th postoperative day.
Figure 3: Peroperative view revealing a ruptured right cornual pregnancy (a) and macroscopic view of the resected specimen (b)

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  Discussion Top

HP was first described by Duverney in 1708 as an autopsy finding.[2] It can occur in different forms: IUP and tubal, abdominal, cornual, cervical, cesarean scar or ovarian pregnancy.[2] Fallopian tube is the most frequent reported site (72%)[3] as was in our patients. Although the ectopic pregnancy was in the right tube in two cases, there is no predilection for right or left fallopian tube.[4] In the general population, HP is a rare entity and occurs in about 1 in 30,000 pregnancies.[2],[5] It mostly happens with known risk factors notably assisted reproductive techniques, ovulation induction, and history of PID or ectopic pregnancy. The risk factors we have found in our cases were ovulation induction in case 2 and previous history of ectopic pregnancy in case 3. However, case 1 was a challenging one as she did not have any of the known risk factors in addition of endometriosis or tubal anomalies (according to peroperative exploration). CC is used to induce ovulation in selected populations with oligo or anovulation as a strategy to increase follicular number and estrogen concentration and could be associated with HP in up to 1/900 of the cases.[6] It is interesting that in case 2, as well as in other cases reported in the literature,[7],[8] the drug had been given from days 2 to 6 of the cycle, a time when the resultant FSH surge is more likely to enhance maturation of multiple follicles. In some previous reports, the drug had been given from days 5 to 9 of the cycle, a time when it is more likely to result in maturation of the dominant follicle only.[6],[9] Preoperative diagnosis of HP is difficult and requires a high index of suspicion. Clinical features can vary widely from asymptomatic to abdominal pain (like our cases) and vaginal bleeding. On ultrasound, the identification of a gestational sac outside the uterus is the gold standard for the diagnosis of ectopic pregnancy. According to reviews, in 77%–85% of cases in whom diagnosis was done through ultrasound scan, diagnosis was made between 5 and 8 weeks of gestation.[6] However, an overwhelming diagnostic difficulty was faced in case 3; an adnexal mass was seen transvaginally with free fluid in the peritoneal cavity and interpreted incorrectly as a ruptured corpus luteum cyst or as a hemorrhagic ovarian cyst along with the presence of an IUP, thinking least of HP. The echogenicity of an adnexal mass may help distinguish the tubal ring of an ectopic pregnancy from corpus luteum and ovarian cyst. In fact, the tubal ring of an ectopic pregnancy is usually more echogenic than ovarian parenchyma, and the corpus luteum is usually equal to or less echogenic than ovary.[10],[11] Plasmatic b-HCG level is often difficult to interpret because the IUP causes the b-HCG level to rise appropriately. Consequently, it was not performed in our cases. Immediate surgical intervention remains the only viable option for acute presentation of HP. The main aim of the surgery should be the preservation of the IUP with minimal manipulation of the uterus. Many cases are treated by surgery through laparoscopy or laparotomy, including salpingotomy or salpingectomy. The choice between conservative and radical treatments may be difficult. A review demonstrated no difference in survival rates of IUP after conservative or radical surgery for tubal ectopic pregnancy.[12] In our cases, lack of laparoscopic equipment in the operating room for emergency, minilaparotomy was performed with salpingectomy for rapid treatment and control of active bleeding. Intrauterine fetal outcome after surgical management depends on many factors: gestational age of diagnosis, operative time, quality of anesthesia, and surgery (minimal uterine manipulation). In case 3, we suggest that the absence of peroperative tocolysis was a factor of IUP abortion on the 2nd postoperative day. Tocolysis was not used due to the absence of fetal pole on ultrasound. The use of prophylactic tocolysis during and after surgery for prevention of uterine contractions still debatable by several authors.[13] In [Table 1], we summarized clinicopathological features of our patients. In situ injections of potassium chloride (KCL) or hyperosmolar glucose can be used if the HP is in early stage. Moreover, since the risks of continued growth and rupture still exist with such nonsurgical management, repeated ultrasound examination and close monitoring of clinical symptoms are essential. A literature review of HP treated with KCL injection noted that 55% of cases failed this therapy and required surgical intervention.[14] Methotrexate is contraindicated in the presence of a viable IUP. Expectant management of HP has also been reported in selected cases; however, a substantially favorable risk-to-benefit ratio should be demonstrated before the adoption of this form of treatment.[15]
Table 1: Cases characteristics

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  Conclusion Top

These cases suggest that confirming an IUP does not exclude the coexistence of an ectopic pregnancy, particularly if ovulation induction has done. Surgical approach with minimal uterine manipulation and prophylactic tocolysis should be preferred to preserve the intrauterine ongoing pregnancy. With early diagnosis and appropriate treatment, 70% of IUP will reach viability.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Talbot K, Simpson R, Price N, Jackson SR. Heterotopic pregnancy. J Obstet Gynaecol 2011;31:7-12.  Back to cited text no. 1
Chawla S, Abulhassan N. An unanticipated case of heterotopic pregnancy. Int J Reprod Contracept Obstet Gynecol 2016;5:1228-30.  Back to cited text no. 2
Barrenetxea G, Barinaga-Rementeria L, Lopez de Larruzea A, Agirregoikoa JA, Mandiola M, Carbonero K, et al. Heterotopic pregnancy: Two cases and a comparative review. Fertil Steril 2007;87:417.e9-15.  Back to cited text no. 3
Han SH, Jee BC, Suh CS, Kim SH, Choi YM, Kim JG, et al. Clinical outcomes of tubal heterotopic pregnancy: Assisted vs. spontaneous conceptions. Gynecol Obstet Invest 2007;64:49-54.  Back to cited text no. 4
Lincenberg KR, Behrman ER, Bembry JS, Kovac CM. Uterine rupture with cesarean scar heterotopic pregnancy with survival of the intrauterine twin. Case Rep Obstet Gynecol 2016;2016:6832094.  Back to cited text no. 5
Mehrabian F, Robati BK. Heterotopic abdominal pregnancy following ovulation induction with clomiphene citrate. Iran J Reprod Med 2010;8:51-4.  Back to cited text no. 6
McClure N, McClelland R, Winter H, White R. Heterotopic pregnancy. J Obstet Gynecol 1991;11:101-3.  Back to cited text no. 7
Selo-Ojeme DO, GoodFellow CF. Simultaneous intrauterine and ovarian pregnancy following treatment with clomiphene citrate. Arch Gynecol Obstet 2002;266:232-4.  Back to cited text no. 8
Naki MM, Tekcan C, Uysal A, Güzin K, Yücel N. Heterotopic pregnancy following ovulation induction by clomiphene citrate and timed intercourse: A case report. Arch Gynecol Obstet 2006;274:181-3.  Back to cited text no. 9
Frates MC, Visweswaran A, Laing FC. Comparison of tubal ring and corpus luteum echogenicities: A useful differentiating characteristic. J Ultrasound Med 2001;20:27-31.  Back to cited text no. 10
Jeri AR. Woman with heterotopic pregnancy after natural conception. BJOG 2015;122:1470-328.  Back to cited text no. 11
Aust T, O'Neill A, Cario G. Purse-string suture technique to enable laparoscopic management of the interstitial gestation of a heterotopic pregnancy. Fertil Steril 2011;95:261-3.  Back to cited text no. 12
Diallo D, Aubard Y, Piver P, Baudet JH. Heterotopic pregnancy: About 5 cases and literature review. J Gynecol Obstet Biol Reprod 2000;29:131-41.  Back to cited text no. 13
Goldstein JS, Ratts VS, Philpott T, Dahan MH. Risk of surgery after use of potassium chloride for treatment of tubal heterotopic pregnancy. Obstet Gynecol 2006;107:506-8.  Back to cited text no. 14
Baxi A, Kaushal M, Karmalkar H, Sahu P, Kadhi P, Daval B, et al. Successful expectant management of tubal heterotopic pregnancy. J Hum Reprod Sci 2010;3:108-10.  Back to cited text no. 15
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