Spontaneous Rupture of Uterine Vessels in the Third Trimester of Pregnancy Resulting in Severe Hemoperitoneum and Fetal Death: A Case Report and Literature Review

Introduction

Spontaneous hemoperitoneum during pregnancy is a rare complication, with significantly high maternal and perinatal mortality rates.¹ The incidence in developing countries is not well known. However, from studies done in Italy and Netherlands, the incidence ranges from 0.004 in 1000 births to 4.9 in 100,000 births, respectively.^2,3 The differential diagnosis includes rupture of utero-ovarian vessels and solid organs, including the spleen and liver.⁴ It occurs most often after the second half of pregnancy and its presentation is sometimes vague. However, common manifestations include abdominal pain and distension associated with derangement of vital signs, anaemia, and signs of fluid accumulation in the peritoneum.⁵ In addition to the clinical findings, investigation like complete blood count and abdomino-pelvic imaging using ultrasound and computerised tomography are essential to reach into diagnosis.

Commonly identified risk factors include endometriosis, multiple pregnancies, advanced age, artificial reproductive technology, and connective tissue disorders.^2,6,7 Although perinatal complications remain high, thorough assessment and a high suspicion index, with adequate replacement of fluid and appropriate emergency surgical interventions, help to decrease maternal complications and mortality.³

Herein, we highlight the importance of early diagnosis and intervention in a rare case of spontaneous hemoperitoneum during the third trimester of pregnancy due to rupture of uterine vessels.

Case Report

A 25-year-old Somalian, primigravid lady at 33 weeks of gestation presented to the emergency department at Daruxannan hospital with complaints of acute abdominal pain and abdominal distention for 16 hours. In addition, she experienced four episodes of non-projectile vomiting and light headedness. She denied abdominal trauma or a chronic personal or family history of medical illness. Upon admission, she was haemodynamically unstable, with a blood pressure of 90/60 mmHg, pulse rate of 110 beats per minute, respiratory rate of 20 breaths per minute, and axillary temperature of 36.5°C. Her oxygen saturation (spO2) was 97%, and Glasgow coma scale of 15/15. Abdominal examination revealed a distended abdomen with significant direct and rebound tenderness, and absent foetal heartbeat. Laboratory investigations revealed elevated white blood cell count with 80% granulocyte count, and a slightly reduced haemoglobin level. Urine analysis also revealed +2 ketones, and +1 proteinuria (Table 1).

Table 1 Laboratory Investigations, a Case of Spontaneous Rupture of Uterine Venous Vessels in the Third Trimester of Pregnancy, Somalia, 2023

After securing initial resuscitative measures with two litres of intravenous normal saline and providing anti-pain, patient was directed for abdominal imaging. Abdominopelvic and obstetric ultrasound showed a singleton non-viable intrauterine pregnancy with a gestational age of 33 weeks and an estimated foetal weight of 2200 gram. Free fluid with echo debris was observed in the peritoneal cavity, including the culde sac, paracolic gutter, and the hepatorenal recess (Figure 1). Otherwise, the solid intra-abdominal organs and the bowel loops were unremarkable.

Figure 1 Abdomino-pelvic ultrasound, 25 years old pregnant lady at 33 weeks gestation with spontaneous hemoperitoneum due to rupture of uterine vessels. Notes: The image labeled (A) showing intrauterine pregnancy, blue arrow showing no colour flow on the fetal heart. The image labelled (B) showing right upper quadrant abdominal ultrasound, white arrow showing free fluid in the hepatorenal recess.

Since the ultrasound finding was inconclusive, the patient was subjected to cross-sectional tomography. Abdominopelvic Contrast-enhanced Computed tomography scan showed significant free fluid in the peritoneal cavity with an average density of 33 HU, uninterrupted uterine wall enhancement, and a well-formed foetus without contrast in the vascular system, indicating non-viability of the foetus (Figure 2). The placenta was observed in the right lateral aspect of the uterus and showed a contrast bushing. No signs of inflammation or wall defects were observed in the bowel. All solid intra-abdominal organs were found to be normal.

Figure 2 Abdomino-pelvic contrast enhanced computerised tomography, 25 years old pregnant lady at 33 weeks gestation with spontaneous hemoperitoneum due to rupture of uterine vessels. Abbreviations: R, right; L, left; S, sagital. Note: CT multiplanar reconstruction image, blue arrow showing intact uterine outlines, white arrow showing intrauterine fetus, green arrow placenta attached to left uterine wall, and red arrow showing intraperitoneal fluid collection.

The patient was diagnosed with spontaneous hemoperitoneum of indeterminate origin. The differential diagnoses considered were ruptured solid intraperitoneal organs and possible hollow organ perforations. Cross-matched fresh whole blood was transfused in addition to two liters of ringer lactate solution. Ampicillin 2 gram intravenous four times a day, Gentamicin 80 mg intravenous three times a day, and Metronidazole 500 mg intravenous three times a day were administered to the patient considering possibility of sepsis. Upon involving a General Surgeon, immediate surgical intervention and an emergency laparotomy were performed. A subumbilical midline abdominal incision was done. Upon entry, 2000 mL of hemoperitoneum was evacuated. A freshly dead foetus was delivered through an incision in the lower uterine segment. After delivery, the uterus was exteriorised, and active bleeding was detected in the ruptured left posterior uterine venous vessel (Figure 3). The ruptured uterine vein was carefully clamped and sutured with a delayed absorbable suture size 0 and round body needle. Bleeding vessels were sutured to ensure proper closure both above and below the bleeding sites. The uterine incision was closed in two layers. After inserting the uterus back and placing an intraperitoneal drain, the abdominal wall was closed layer-by-layer. Three units of cross-matched fresh whole blood were administered intraoperatively.

Figure 3 Intraoperative finding, 25 years old pregnant lady at 33 weeks gestation with spontaneous hemoperitoneum due to rupture of uterine vessels. Note: Intraoperative finding, white arrow showing the site of the ruptured uterine venous plexus and blue arrow showing left uterine cornua.

Post operatively, the patient continued intravenous antibiotics; maintenance fluid with Ringer lactate solution was continued; and she was strictly followed for vital sign, urine output, and abdominal conditions. Her haemoglobin on her second day of operation was 13grams/dl and her white blood cell count was 1500/µl with 75% granulocyte. Her recovery period was smooth. She was discharged after five days of in-hospital stay and her subsequent follow-ups were uneventful.

Discussion

Spontaneous hemoperitoneum in pregnancy due to rupture of uterine vessels is an unusual case that presents a risk of maternal and foetal death. Several reports have shown spontaneous rupture of the uterine vessels during pregnancy, particularly during the third trimester and puerperium.^8–11 Our case report also showed the appearance of this extremely unusual condition in the third trimester of pregnancy, with foetal death and life-threatening maternal complications.

Several risk factors for spontaneous rupture of uterine vessels have been identified during pregnancy, including endometriosis, artificial reproductive technology, connective tissue disease, and elevated venous pressure, especially during labour.^2,6,7 In our case, a significantly tortuous utero-ovarian venous plexus was identified, which may have contributed to this condition.

Typically, pregnant patients with spontaneous hemoperitoneum experience severe abdominal pain and signs of hypovolaemia. Similarly, our patient presented with abdominal pain associated with hemodynamic instability, evidence of intraabdominal fluid collection, and no obvious history of trauma or vaginal bleeding. Imaging, including abdominal ultrasound and contrast CT scan with contrast, is extremely important for detecting free fluid in the abdomen and ruling out potential causes, including uterine rupture, solid abdominal organ rupture, perforated viscous, and appendicitis.^2–4,12 However, owing to the vague patient presentations and rare occurrences, a high index of suspicion is particularly important for diagnosis.

Cases of spontaneous rupture of the uterine venous plexus with insidious onset and different clinical manifestations have been reported. A case report illustrated that a pregnant woman in her first pregnancy at 33 weeks of gestation presented with signs and symptoms of preterm labour and was treated with tocolytics, analgesics, and steroids for lung maturation. Despite extensive investigations, the cause of the patient’s presentation remains unknown. However, during undergoing caesarean section, the patient had a spontaneous uterine venous plexus and ovarian rupture.¹⁰ In contrast to the aforementioned case, our patient presented with acute and life-threatening complications in the mother and intrauterine foetal death. This contrasts with the stable hemodynamic condition and favourable foetal outcomes observed in other cases.

In a different case report, a 32-year-old mother in her third pregnancy at 16 weeks of gestation presented severe abdominal pain associated with hypovolemic shock after spontaneous rupture of the uterine vessels was diagnosed using abdominal imaging, and an immediate surgical intervention with ligation of the ruptured vessel was performed.¹³ Although the presentation of the case was acute and similar to that of our case, the pregnancy was viable, occurred in the early second trimester of pregnancy, and was managed to reach term with a subsequent good foetal outcome. In our case, the foetus was nonviable, and delivery was performed during laparotomy. Most importantly, early detection and management are essential to optimise outcomes for both the mother and the foetus.

The clinical approach and treatment of acute abdominal syndrome during pregnancy pose diagnostic and management challenges. Prioritisation during evaluation should be performed to detect patients with a life-threatening cause of their symptoms and to identify those who require immediate surgical exploration. Most importantly, spontaneous hemoperitoneum cases during pregnancy, prompt diagnosis, and immediate replacement of intravascular volume with urgent surgical intervention are invaluable in reducing the high maternal and perinatal mortality associated with this rare condition. Finally, addressing all potential causes and risk factors contributes to decreasing recurrence and optimising subsequent pregnancy outcomes (Table 2).

Table 2 Literature Review of Case Reports of Spontaneous Rupture of Uterine Vessels in Pregnancy

Conclusion

Spontaneous rupture of the uterine vessels during pregnancy can lead to severe hemoperitoneum and foetal death. Usually, patients will present with sudden onset of abdominal pain, shock, and distended tender abdomen. A CT of the abdomen and pelvis can help in diagnoses. These cases require urgent surgical intervention to avoid complications. Ideally, exploratory laparotomy with suturing of ruptured uterine vessels should be done.