Early abdominal pregnancy in a spontaneous heterotopic pregnancy: Case report

Abstract

Introduction and significance

Spontaneous heterotopic pregnancies, concurrently occurring intrauterine and ectopic pregnancies, pose a substantial risk to maternal health and are often misdiagnosed. This case report details the challenges in identifying and managing an exceptionally rare case of abdominal pregnancy without assisted reproduction. The patient's initial misdiagnosis underscores the complexities in diagnosis, emphasizing the importance of comprehensive imaging techniques.

Case presentation

We present the case of a 36-year-old gravida 5, para 3, with a history of dilation and curettage, experiencing a heterotopic pregnancy involving delayed miscarriage in both uterine and abdominal cavities. Despite presenting symptoms of pelvic pain and abnormal vaginal bleeding, the abdominal pregnancy was initially overlooked in ultrasound examinations. The accurate diagnosis was only achieved post-miscarriage, leading to a timely intervention through laparotomy.

Clinical discussion

The absence of identifiable risk factors, except for the patient's history of dilation and curettage, highlights the spontaneous nature of this non-assisted reproduction-related pregnancy. This case emphasizes the challenges in diagnosing and managing spontaneous heterotopic pregnancies, particularly when an abdominal pregnancy is involved. Vigilance and advanced imaging techniques are crucial for early recognition and appropriate intervention.

Conclusion

This unique case underscores the difficulties in diagnosing and managing spontaneous heterotopic pregnancies, especially when an abdominal pregnancy is present. Vigilance and advanced imaging are essential to identify rare occurrences like abdominal pregnancies that may go unnoticed in conventional ultrasound examinations. Early recognition and intervention are critical in averting potential life-threatening consequences associated with this uncommon condition.

Highlights

• •Unusual pregnancy combination
• •Diagnostic oversight and delayed recognition
• •Complexities in diagnosis and surgical management
• •Diagnostic approaches and imaging significance

1. Introduction

A heterotopic pregnancy refers to the simultaneous occurrence of an intrauterine pregnancy and an ectopic pregnancy within the same pregnant woman. Primary abdominal pregnancy is considered one of the most uncommon forms of ectopic gestation, with an unknown incidence rate [1]. Both primary and secondary abdominal pregnancies, collectively reported at 1 in 10,000 cases, manifest symptoms such as abdominal pain or spotting, often detectable through sonography. However, some cases escape diagnosis, and improper management can lead to severe complications.

The risk factors for heterotopic pregnancy include the increased use of assisted reproductive technology, previous tubal surgery, and the presence of hydro-salpinx. In this context, we present a case of heterotopic pregnancy that went undiagnosed, resembling perinatal care for a delayed miscarriage [2]. The primary diagnosis was established during the operation, revealing a rare combination of spontaneous heterotopic pregnancy and abdominal pregnancy, wherein the intrauterine pregnancy resulted in an early miscarriage [3].

This work was reported in line with the SCARE criteria [12].

2. Observation: a case study

A 36-year-old female conceived spontaneously. She is gravida 5, para 3, she have a history of dilation and curettage. Her last menstrual period was 12 weeks before presentation, and a intrauterine delayed miscarriage pregnancy (6 weeks of gestation) had been diagnosed by her gynecologist by means of ultrasonography. In fact, she have heterotopic pregnancy a delayed miscarriage pregnancy which is in intra uterin and the other in the abdominal cavity. Her pregnancy was initially misdiagnosed and managed as miscarriage pregnancy at 6 weeks of pregnancy. She was diagnosed at 6 weeks gestation with a singleton intrauterine pregnancy using ultrasound echography. Her gynecologist confirm that she had missed miscarriage because there is a hypotonic gestational sac in the cavity of the uterus. She had spontaneous miscarriage two days after the diagnosis of delayed miscarriage. She has not undergone a curettage.

On the ultrasound negative we find a gestational sac with two hyperechoic rings, the localisation is not centrally inside the uterine cavity. The shape is not ovoid. The diameter is 20 mm (Fig. 1). Her gynecologist didn't saw the abdominal pregnancy in the ultrasound echography. The correct diagnosis was only made after miscarriage. The patient was referred ten days after that to our emergency department. It was suspected that there was an intrauterine pregnancy combined with an abdominal pregnancy based on sonographic findings. She was hospitalized. She complains for persistent pelvic pain and abnormal vaginal bleeding 10 days after miscarriage. The patient denied a history of assisted reproduction and uterine surgery except one dilation and curettage. She also denied a history of pelvic inflammatory disease (PID), endometriosis, cigarette use, and alcohol, cocaine consumption.

Fig. 1.

Ultrasound negative showing that she had missed miscarriage at 6 weeks of pregnancy.

At admission, clinical examination revealed that she was hemodynamical stable, she had a pelvic tenderness, an abnormal vaginal bleeding. She gives us an ultrasound negative showing that she had missed miscarriage at 6 weeks of pregnancy (Fig. 1). At trans-abdominal and endovaginal ultrasonography, we found an empty uterine cavity because the patient had a spontaneous miscarriage; and a 12 weeks pregnancy outside the uterus. It was a evolutive pregnancy. A hemoperitoneum is found too. (Fig. 2) Blood analysis showed a hemoglobin level of 10, 8 g/dl. Blood type was O positive. Clinical status of the patient revealed an emergency condition, so a decision to perform a laparotomy was made. At the surgical procedure we find a hemoperitoneum, enlarged right fallopian tube and the 12 weeks pregnancy foetus and his trophoblast was implanted into the pouch of Douglas. (Fig. 3, Fig. 4, Fig. 5, Fig. 6). The hematic mass, including the foetus and chorionic villi was extracted from peritoneum and hemostasis was performed. There was not a major complications after surgery. The mother was discharged in good health 5 days after surgery. We performed a weekly monitoring for the serum β-HCG level until negativity.

Fig. 2.

Endovaginal ultrasonography: empty uterine cavity and 12 weeks abdominal pregnancy.

Fig. 3.

Laparotomy: hemoperitoneum.

Fig. 4.

Abdominal pregnancy with enlarged right fallopian tube.

Fig. 5.

12 weeks pregnancy foetus with trophoblast.

Fig. 6.

intraoperative ectopic pregnancy.

Our conclusion was early (≤20 weeks of gestation) secondary abdominal pregnancy.

3. Discussion

Encountering a case of abdominal pregnancy is a rare occurrence in the career of most gynecologists, with an incidence reported at approximately 1 in 10,000 live births and 9.2 per 1000 ectopic pregnancies [1]. The instance described in this report is categorized as a secondary abdominal pregnancy, signifying that the embryo initially implanted in the fallopian tube, then extruded or expelled, and subsequently implanted itself on another intra-abdominal surface [4].

Studdiford's criteria for diagnosing primary abdominal pregnancy involve the presence of normal bilateral tubes and ovaries with no evidence of recent or past pregnancy, the absence of a uteroperitoneal fistula, and the exclusive connection of the pregnancy to the peritoneal surface, early enough to eliminate the possibility of secondary implantation after primary tubal nidation. Primary peritoneal pregnancy has also been associated with foci of endometriosis. Another classification relevant to diagnosis and treatment distinguishes early peritoneal pregnancy (≥20 weeks of gestation) from advanced cases (≤20 weeks of gestation) [5].

Diagnosing abdominal pregnancy poses a challenge, as symptoms are nonspecific, including abdominal pain, vaginal bleeding, tenderness upon palpation, and palpable fetal parts in advanced cases [6].

According to one study, the symptoms reported were very different, with an overwhelming majority of patients were asymptomatic at the time the heterotopic pregnancy was discovered [7]. Pre-operative confirmation of diagnosis necessitates abdominal ultrasound examination, with key diagnostic findings being an empty uterine cavity separate from the foetus and the presence of an abdominal ectopic placenta [8]. The presence of intrauterine gestational sac doesn't exclude the presence of ectopic pregnancy.

Historically, abdominal pregnancies were often diagnosed late, but advancements in diagnostics, such as serial β-HCG, high-quality ultrasonography, and MRI, have facilitated earlier detection. MRI proves useful in confirming abdominal pregnancy, identifying the site of placenta attachment, and assessing the vascular supply and integrity of the gestational sac [9].

Despite these diagnostic improvements, cases of this rare condition continue to go undiagnosed, leading to delayed diagnoses made during laparotomy [10].

The management of abdominal heterotopic pregnancy is even harder. In fact surgical management of advanced abdominal pregnancy, involving the removal of the ectopic placenta, carries risks such as massive bleeding, hypovolemic shock, and the need for intra-operative blood transfusions, potentially leading to serious complications [11]. Alternatively, a conservative approach, leaving the placenta in situ after fetal removal, has been proposed as a safe option [12]. The described experience in this report suggests successful outcomes, possibly attributed to the early nature of the abdominal pregnancy [13].

The surgery is standard of care for treatment of abdominal and the management can be by laparotomy or laparoscopy. The monitoring of for the serum β-HCG level until negativity seems mandatory after surgery. In case of stagnation of serum β-HCG level, we need to prescribe methotrexate as a medical adjuvant of surgery.

One other case of term spontaneous heterotopic pregnancy (Abdominal and Intrauterine) was been reported and the diagnosis was made after cesarean section. Her pregnancy was initially misdiagnosed and managed as a di-amniotic di-chorionic gestation [14].

Our case is unique and different from other cases because it was about not advanced abdominal pregnancy, the maternal outcomes and morbidities were not complicated.

However, like all the other cases, there was been a misdiagnosis at the first consultation despite the experience and the good reputation of the referring gynecologist.

4. Conclusion

Ultrasonic examination is the first step to diagnose heterotopic pregnancies especially for the patients who had many risk factor in their medical history. Therefore, careful extrauterine examination is necessary and finding a gestational sac in uterine cavity is not cause for finalizing examination. Diagnosis may yet be challenging because the presence of an intrauterine pregnancy falsely reassures most clinicians. Any doubt in diagnosis should be confirmed with other imaging modalities.

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.

Availability of supporting data

Not applicable.

Ethical approval

Not applicable. Our institution requires no ethical approval for case reports.

Funding

Not applicable.

All authors read and approved the final manuscript.

Author contribution

Marzouk Khouloud, conception and design, acquisition of data drafting the article, revising it critically for important intellectual content final approval of the version to be published.

Othman Maroua, conception and design, acquisition of data drafting the article, revising it critically for important intellectual content final approval of the version to be published.

Hafsi Montacer, conception and design, acquisition of data drafting the article, revising it critically for important intellectual content.

Swileh Salma, final approval of the version to be published.

Smida Safa, final approval of the version to be published.

Bouchahda Haifa, final approval of the version to be published.

Guarantor

The corresponding author is the guarantor of submission.

I, Montacer Hafsi hereby affirm that this manuscript is original, has not been published previously, and is not under consideration for publication elsewhere. All authors have contributed significantly to this work and have reviewed and approved the final version of the manuscript for submission. We confirm that all sources used have been properly cited and that any conflicts of interest have been disclosed.

Declaration of competing interest

All authors declare that they have no conflicts of interest.