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

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 Lists of Investigation Results Reference Range Haemoglobin 10.1g/dl 12–16 g/dl White blood cells 30,900/ul 4000–10,000/ul Platelet 351,000/ul 150,000–450,000/ul Aspartate aminotransferase 30 U/L 0–42 U/L Alanine aminotransferase 35 U/L 0–37.9 U/L Creatinine 1.2 mg/dl 0.5–1/1 mg/dl Blood urea nitrogen 40mg/dl 10–50 mg/dl Urine Analysis +proteinuria and +ketoneuria Not available 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. Abdomino-pelvic ultrasound, 25 years old pregnant lady at 33 weeks gestation with spontaneous hemoperitoneum due to rupture of uterine vessels. 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. 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. Abbreviations : R, right; L, left; S, sagital. Abdomino-pelvic contrast enhanced computerised tomography, 25 years old pregnant lady at 33 weeks gestation with spontaneous hemoperitoneum due to rupture of uterine vessels. 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. Intraoperative finding, 25 years old pregnant lady at 33 weeks gestation with spontaneous hemoperitoneum due to rupture of uterine vessels. 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.

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.

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.

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 Author, Year Age Parity Mode of Conception Number of Gestations GA (Weeks) Ruptured Vessel Clinical Presentation Diagnostic Modality Surgical Intervention Complications Follow Up Dogeret al, 2013 8 26 Nulliparous IVF Twin 32 weeks Utero-ovarian vein Abdominal pain Clinical Laparotomy, C/S, Uterine vein branch sutured PTB Un eventful Andrés-Orós et al, 2010 9 30 Parous Natural Twin 33 Uterine vein Abdominal pain Clinical Uterine Vein Sutured, C/S IUFD Un eventful Ruan et al, 2023 10 24 Nulliparous Natural Singleton 33 Uterine venous combined with ovarian rupture Lower abdominal pain Abdominal Ultrasound and US guided abdominal Puncture, Exploratory Laparotomy Exploratory Laparotomy, ligation of bleeding points, CS PTB Uneventful Wanget al, 2021 11 32 Nulliparous Natural Singleton Post partum Left Uterine vein Signs of shock, abdominal pain and tenderness Abdominal Ultrasound, abdominal Puncture Exploratory laparotomy, suturing of the ruptured vessel DIC, Massive blood transfusion Uneventful Jayalath et al, 2021 13 32 Parous Natural Singleton 16 weeks Uterine artery Acute Abdominal pain, Signs of hypovolemia Abdominal Ultrasound Emergency Laparotomy, Suturing of ruptured artery None Her pregnancy was continued until 36 weeks Hamedah etal, 2017 14 38 Parous Natural Singleton 38 Left utero-ovarian venous plexus Abdominal pian Clinical diagnosis Exploratory laparotomy, ligation of bleeding vessels, caesarean section None Uneventful Lim et al 2016. 15 24 Parous Natural Singleton 37 Venous plexus in the posterior wall of the uterus Lower abdominal pain Clinical diagnosis and abdominal ultrasound Exploratory laparotomy, ligation of bleeding vessels, caesarean section None Williamson et al 2011 16 37 Parous Natural Singleton Postpartum Left uterine vein Abdominal pain and Tenderness Clinical diagnosis and CT scan of the pelvis Spontaneous vaginal delivery and conservative management None Follow up MRI after 4 months showed severe endometriosis Roche et al, 2009 17 32 Parous IVF Twin 33 Right uterine vessels Acute abdominal pain and hypovolemic shock Abdominal Ultrasound and exploratory laparotomy Exploratory laparotomy and dissection of the uterus None Uneventful Zhang et al, 2009 18 38 Parous IVF Twin 29 Uterine varix on the posterior surface of uterine wall Acute abdominal pain, anaemia CT scan and abdominal paracentesis Exploratory laparotomy, ligation of bleeding vessels, caesarean section IUFD Uneventful Zhang et al, 2009 18 35 Parous IVF Single 35 Right side uterine veins Acute abdominal pain, and hypovolemic shock Clinical diagnosis, Exploratory laparotomy Exploratory laparotomy, ligation of bleeding vessels, caesarean section Massive transfusion Uneventful Zhang et al, 2009 18 34 Parous IVF Single 30 Left corneal vein Acute abdominal pain, and hypovolemic shock Abdominal ultrasound and Exploratory laparotomy Exploratory laparotomy, ligation of bleeding vessels, and caesarean section Massive transfusion and neonatal asphyxia Uneventful Our case 25 Nulliparous Natural Singleton 33 Left posterior uterine venous plexus Acute abdominal pain, and hypovolemic shock Abdominal ultrasound, Computerized Tomography scan, and Exploratory laparotomy Exploratory laparotomy, ligation of bleeding vessels, and caesarean section Blood transfusion, IIUFD Uneventful Abbreviations : IUFD, Intra Uterine Fetal Death; CT, Computerised Tomography; GA, Gestational Age; DIC, Disseninated intravascular Coagulation; PTB, Preterm Birth. Literature Review of Case Reports of Spontaneous Rupture of Uterine Vessels in Pregnancy Abbreviations : IUFD, Intra Uterine Fetal Death; CT, Computerised Tomography; GA, Gestational Age; DIC, Disseninated intravascular Coagulation; PTB, Preterm Birth.