The unfortunate twin: A case report of intrapartum diagnosis of heterotopic pregnancy.

A 32-year-old woman, gravida 2 para 1 (previous spontaneous vaginal delivery), presented at 40 weeks of gestation, confirmed by a 10-week ultrasound, after an uneventful antenatal course. She had conceived naturally and had no history of sexually transmitted diseases or pelvic inflammatory disease. She had had no previous abdominal or tubal surgeries, and had no smoking history, and no history of ectopic pregnancy. The ultrasound scan at 10 weeks showed a crown–rump length (CRL) indicative of 10 weeks with positive fetal heart tone. The adnexa was not examined well as the patient was asymptomatic. A fetal anatomic survey was done at 22 weeks and biophysical profile was done at 34, 36 and 38 weeks where the patient was asymptomatic with no complaint throughout the pregnancy. The patient arrived in labor after five hours of contractions and diffuse abdominal pain, but denied any danger signs such as vaginal bleeding, fluid loss, or vomiting. On examination, she was in active labor with stable vital signs (BP 120/70 mmHg, pulse 96 bpm), pink conjunctiva, and a 40-week gravid uterus, with three strong contractions every 10 min. The fetus was in longitudinal lie and cephalic presentation and the fetal heart rate was 144 bpm. Abdominal palpation revealed diffuse tenderness, most pronounced in the right lower quadrant. Pelvic exam showed a cervix dilated to 4 cm, 100 % effaced, at station 0. Artificial rupture of membranes revealed grade 2 meconium-stained amniotic fluid. Ultrasound identified minor free fluid in the peritoneum. Cesarean delivery was indicated due to latent first stage of labor and meconium-stained fluid. Intraoperatively, 600 ml of hemoperitoneum was found, along with an intact gravid uterus and well-formed lower uterine segment. A healthy male neonate was delivered (4.3 kg, APGAR scores of 8 and 9 at one and five minutes). Further exploration revealed active bleeding from the right tubal ostium and an intact 5 × 4 cm ampullary ectopic pregnancy. Right salpingectomy was performed, and histopathology confirmed ectopic pregnancy. (see Fig. 1 , Fig. 2 ) The patient's postoperative course was uneventful, and she was discharged on the third day in stable condition. Fig. 1 Intraop picture of the intact gravid uterus (blue arrow), tubal ectopic pregnancy (green arrow), and active ostial bleeding (black arrow). Fig. 1 Fig. 2 A picture of the tube after salpingectomy with the tubal ectopic pregnancy (yellow arrow). Fig. 2 Intraop picture of the intact gravid uterus (blue arrow), tubal ectopic pregnancy (green arrow), and active ostial bleeding (black arrow). A picture of the tube after salpingectomy with the tubal ectopic pregnancy (yellow arrow).

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Written informed consent was obtained from the patient for publication of this case report and accompanying images.

Heterotopic pregnancy is a rare but potentially life-threatening condition requiring high clinical suspicion for timely diagnosis, especially in patients treated with assisted reproductive technologies. First-trimester ultrasound scanning should thoroughly investigate the adnexa to identify potential extrauterine pregnancies. Transvaginal ultrasound is key for detection. Management should focus on removing the ectopic pregnancy while preserving the intrauterine pregnancy, with surgery being the preferred approach in most cases. Early diagnosis and appropriate treatment are essential for favorable maternal and fetal outcomes.

Heterotopic pregnancy is the presence of an intrauterine pregnancy and an ectopic pregnancy at the same time [ 1 , 5 ]. The pregnancy can rarely be triplet or even quadruplet, though with few reports of the latter [ 6 ]. The commonest site for the ectopic pregnancy is the tube, although ovaries, peritoneal cavity, and cervix are also possible [ 6 , 7 ]. The reported incidence of heterotopic pregnancy following natural conception is around 1:30,000 pregnancies, but its incidence is higher in pregnancies with assisted reproductive technologies, reaching up to 1:100. The overall incidence is around 1:3600 [ 5 , 7 , 8 ]. Most of the risk factors for heterotopic pregnancy are similar to those for ectopic pregnancy. These include previous ectopic pregnancy, history of previous tubal or abdominal surgeries, pelvic inflammatory disease, intrauterine device placement, endometriosis, and smoking [ 3 , 5 , 9 , 10 ] The use of assisted reproductive technology (ART) significantly increases the risk of having a heterotopic pregnancy. The reasons behind this include the number of embryos transferred to the uterus. Additionally, one of the indications for ART is tubal pathology, which increases the odds of having heterotopic pregnancy. It should be noted that many patients might not have any risk factor [ 5 , 6 , 11 , 12 ]. The patient presented here did not have any of the risk factors mentioned. Patients with heterotopic pregnancy can present with several symptoms, the main ones being lower abdominal pain, amenorrhea, and vaginal spotting but some patients are asymptomatic, especially during early gestation [ [4] , [5] , [6] , 10 ]. Most symptoms begin from the 5th to 8th week of gestation, although there are reports of asymptomatic cases in the second and third trimesters. After rupture of the tubal pregnancy, hemoperitoneum will develop, leading to syncope, vomiting, hemorrhagic shock, and loss of consciousness. Due to the presence of a viable intrauterine pregnancy, the symptomatology is usually attributed to a threatened or ongoing abortion [ 2 , 3 , 5 , 12 ]. The patient in the case reported here presented with lower abdominal pain and tenderness while laboring. She most likely had a missed quiescent tubal pregnancy, which was conceived at the same time as the intrauterine pregnancy. The mechanical and biochemical factors of labor, like prostaglandins, have initiated the bleeding intrapartum despite the silent antepartum period. Transvaginal ultrasound is the first-line investigation for suspected heterotopic pregnancy. MRI adds value in cases where some uncertainty remains. Laparoscopy or explorative laparotomy also aids in diagnosis and management [ 5 , 7 , 9 ]. The diagnosis of heterotopic pregnancy is not straightforward. The presence of a viable intrauterine pregnancy usually hinders the search for an extrauterine pregnancy. Additionally, tubal pathologies like cysts can increase the difficulty of visualizing the tubal ectopic pregnancy [ 5 ]. Management is targeted at removing the ectopic pregnancy while maintaining the intrauterine pregnancy [ 2 , 4 ]. There are conservative, surgical, and medical management options. Conservative management is usually done after admitting the patient for close follow-up, to intervene if things worsen. Around 60 % of patients undergo spontaneous resorption of the extrauterine pregnancy, while around 20 % experience tubal rupture during conservative management [ 3 ]. Conditions to be considered for expectant management are gestational age of less than 7 weeks and size of the ectopic fetus less than 3.5 cm. If the patient does not fulfill the conditions for conservative management, surgical intervention should proceed. Whenever possible, surgical management should be avoided due to anesthesia-related complications for the fetus, such as hydrocephalus and microcephaly [ 7 , 13 ]. Surgical removal of the ectopic pregnancy remains the gold standard management. It can be either laparoscopic or an open laparotomy to perform salpingectomy, oophorectomy, or salpingostomy [ 5 , 6 ]. During the procedure, manipulation of the uterus should be kept to a minimum to avoid abortion of the intrauterine pregnancy [ 8 , 10 , 12 ]. Although inconsistent, reports suggest that laparoscopy may have more adverse effects on the pregnancy due to trauma from trocar placement, higher intra-abdominal pressure, and increased CO₂ resorption by the fetus [ 7 ]. Apart from surgical intervention, medical options should be considered, although they are not usually practiced. They include fetal intrathoracic injection of potassium chloride or hyperosmolar glucose under ultrasound guidance. However, methotrexate is contraindicated [ 3 , 6 , 8 , 12 ].

This article was not commissioned and was peer reviewed.

Mesfin Ayalew Tsegaye contributed to the conception of the case report, acquiring and interpreting the data, drafting the manuscript, undertaking the literature review, and revising the article critically for important intellectual content. Meles Tazeb Teloye contributed to patient care, the conception of the case report, drafting the manuscript, undertaking the literature review, and revising the article critically for important intellectual content. Both authors approved the final submitted manuscript.

Heterotopic pregnancy is the occurrence of an intrauterine pregnancy with concomitant extrauterine pregnancy (ectopic pregnancy) [ 1 ]. It is rare, especially following natural conception, although it is more frequent in cases of assisted reproductive technologies or ovarian stimulation [ 2 , 3 ]. Its rarity and the presence of an intrauterine pregnancy make the diagnosis of heterotopic pregnancy difficult. It depends on the clinical symptomatology and transvaginal ultrasound, which might demonstrate an ectopic pregnancy with a fluid collection in the cul-de-sac in addition to the intrauterine pregnancy [ 3 , 4 ]. Most heterotopic pregnancies are diagnosed in the first trimester, although there are rare reports of diagnosis in the second or third trimester [ 5 ]. This report presents a case of tubal ectopic pregnancy incidentally diagnosed at term labor.

The authors declare that they have no conflict of interest regarding the publication of this case report.