Spontaneous hemoperitoneum in the second trimester of pregnancy with a favorable outcome: a case report

Abstract

Spontaneous hemoperitoneum in pregnancy is extremely rare but may cause severe hemorrhage, resulting in a high rate of mortality. We report a case of a primigravid woman who complained of middle-upper abdominal pain. She was admitted to our department at 24 + 6 weeks of in vitro fertilization gestation with a surgical history of endometriosis. The patient’s non-specific presentation posed diagnostic difficulties and intestinal obstruction was initially suspected. Emergent computed tomographic findings showed massive free abdominal fluid, which was suspected to have arisen from the left ovary or uterus. During an emergency exploratory laparotomy, she had interrupted suturing of a ruptured left uterine subserosal vein performed. Computed tomographic imaging is more helpful for isolating the source of bleeding than ultrasound. Prompt action of an exploratory laparotomy can result in favorable outcomes for the mother and newborn in spontaneous hemoperitoneum in pregnancy.

Introduction

Spontaneous hemoperitoneum in pregnancy (SHiP) is a rare but life-threatening complication of pregnancy. SHiP is defined as spontaneous (nontraumatic) intraperitoneal hemorrhage during pregnancy and up to 42 days postpartum, requiring surgical intervention or embolization, and excluding ectopic pregnancy, uterine rupture, and cesarean section-associated bleeding. ¹ The exact incidence of SHiP is unknown because many studies to date have been case reports. The incidence of SHiP was reported to range from 0.004 in 1000 births to 4.9 in 100,000 births in studies from Italy and the Netherlands.^2,3 The perinatal mortality rate of SHiP can be as high as 30%.^4,5 SHiP has been reported in various contexts, including ruptures of the splenic artery, uterine arteries, varicose veins, or aneurysms. Recently, systematic reviews showed that endometriosis and in vitro fertilization were the main risk factors for SHiP.^4–6 We report a rare case of spontaneous rupture of a uterine-surface variceal vessel in the second trimester of pregnancy that mimicked intestinal obstruction and posed diagnostic difficulties and challenges in management. This report aims to share the unique experience of the diagnosis and management of this uncommon emergency. The reporting of this study conforms to the CARE guidelines. ⁷

Case report

A primigravid woman, 24 weeks and 6 days of gestation, visited the emergency room of Shanghai Songjiang District Central Hospital in November 2023, complaining of middle-upper abdominal pain of 16 hours’ duration. The patient had a surgical history of endometrioma in the left ovary, and she used artificial reproductive technology for the current pregnancy. Additionally, she had habitual constipation and a poor appetite during pregnancy. Her pregnancy was followed up regularly. Scans performed during pregnancy did not show any obvious abnormalities. On admission, her vital signs were within the normal range, with a temperature of 36.5°C, pulse of 88 beats/minute, blood pressure of 92/69 mmHg, and respiratory rate of 20 breaths/minute. She denied any trauma or recent coitus before her current presentation. The patient complained of aggravated constipation in the past 4 to 5 days. Palpation of the abdomen showed irregular uterine contractions and intense upper abdominal tenderness with no guarding or rebound. The pain increased with movements and decreased at rest. The fundal height was 24 cm. No pathology was found by a vaginal examination, with no vaginal bleeding, no rupture of membranes, and the cervix was long and closed. An abdominal ultrasound showed a viable fetus in cephalic presentation and a normal placenta with no signs of abruption. Intrauterine bleeding was not present. Free fluid was observed at the inferior edge of the spleen (depth of 51 mm) and liver (depth of 48 mm). Cardiotocography showed a baseline heart rate of 120 beats/minute, with no deceleration. The total white blood cell count was 23.04 × 10⁹/L on admission, and the C-reactive protein (CRP) concentration was 0.001 g/L. The hemoglobin concentration was 103 g/L on admission. The albumin concentration was 29.8 g/L. To exclude appendicitis, an ultrasound of the right lower abdomen was performed, which indicated pneumatosis of the colon. The initial working diagnosis was pregnancy with intestinal obstruction combined with hypoproteinemia. Oral paraffin oil of 50 mL thrice a day was prescribed to lubricate the intestines and relieve intestinal obstruction. To exclude other diagnoses, the patient consented to have emergent computed tomography (CT) of the abdomen and pelvis performed. CT showed massive free abdominal fluid, which was suspected to have arisen from the left ovary or uterus (Figure 1). The patient complained of intensified abdominal pain after the CT scan. She was promptly advised to undergo an immediate exploratory laparotomy. Approximately 6 hours after her admission, the hemoglobin concentration was decreased to 62 g/L. Twenty minutes later, the patient had an exploratory laparotomy performed.

Figure 1.

Computed tomographic images of the abdomen and pelvis show a large volume hemoperitoneum with a mean computed tomographic value of 75 (white arrows). (a): Horizontal view and (b): coronal view.

Under general anesthesia, a midline grid-iron incision was made. Intraoperatively, a hemoperitoneum was found and the uterus was unruptured. A long and narrow blood clot was attached to the anterior uterus. Massive blood clots were noted predominantly along the left parametrial region to the spleen, with a distorted and fragile appearance of the left adnexal veins. At the beginning of the operation, the source of bleeding was not immediately evident. Further exploration after moving the uterus showed a ruptured left uterine subserosal vein with active bleeding (Figure 2). Adequate hemostasis was achieved by interrupted suturing of the bleeding vein. Intraoperative blood loss was estimated at approximately 2400 mL. The patient received 4 units of packed red blood cells and 400 mL fresh frozen plasm. After the laparotomy, intravenous infusion of 15 g of magnesium sulfate was applied in a timely manner to prevent premature birth and protect nerves in the fetal brain. The same dose of magnesium sulfate was administered 24 hours later. On the 10th postoperative day, laboratory results showed that the hemoglobin concentration was 105 g/L. She was discharged from the hospital 12 days after the surgery. Her postoperative recovery was satisfactory. She underwent a successful cesarean delivery at 38 weeks and 3 days of gestation. The mother and her newborn were discharged home on the 5th postoperative day with no morbidity.

Figure 2.

Intraoperative findings. Active bleeding in the subserosal vein (white arrow) of the uterus after moving the uterus.

Discussion

We present a case of life-threatening SHiP due to rupture of the uterine vessels in a singleton pregnancy at 24 weeks of gestation. Several risk factors for spontaneous rupture of the uterine vessels, such as endometriosis, artificial reproductive technology, connective tissue disease, and elevated venous pressure, have been identified during pregnancy.^2,4,8,9 The incidence of SHiP in severe endometriosis is approximately 0.4%.^6,10,11 Additionally, endometriosis is present in 55.9% of patients of SHiP. ¹² In our case, the patient had some of the risk factors for SHiP, namely endometriosis and artificial reproductive technology. The pathophysiology of endometriosis-associated SHiP remains unknown. However, decidualization of an ectopic endometrial stroma, vascular walls, and peritoneal endometriosis sites can be involved in the pathogenesis of SHiP. ⁵ The utero-ovarian vessels are fragile because of decidualization and chronic inflammation associated with endometriosis. During pregnancy, there is an increase in venous pressure in the utero-ovarian circulation. The anatomical changes of pregnancy, in combination with the adhesions caused by endometriosis, most likely have the potential to tear these vessels.^4,13 All of the above-mentioned factors may be responsible for vascular instability and bleeding. Most reported cases of SHiP refer to the involvement of left-sided uterine vessels, similar to our case, possibly because of the combination of natural dextro-rotation of the uterus during pregnancy and the more common left position of the fetal head.^4,14,15 A limitation of our study is the lack of a histopathological examination because specimens were not collected during surgery.

The predominant symptoms of SHiP are acute abdominal pain, flank pain, and fetal heart rate deceleration. Suspecting SHiP on the basis of these nonspecific symptoms until the patient develops hypovolemic shock is difficult.^12,16 The differential diagnosis of SHiP includes placental abruption, uterine rupture, heterotopic pregnancy, HELLP syndrome, severe pre-eclampsia, acute fatty liver disease, acute appendicitis, chorioamnionitis and ruptured liver, spleen or its vasculature.^4,13,17 A delay in diagnosing of SHiP could lead to a catastrophic outcome. Therefore, a timely diagnosis is challenging but important. In our case, intestinal obstruction was suspected because the patient initially had unremarkable abdominal pain with gastrointestinal symptoms and laboratory evidence of infection.

The use of imaging is essential for the diagnosis of SHiP. Ultrasound is the first line of detection of SHiP, and it is helpful in identifying the presence of free peritoneal fluid. However, a CT scan or magnetic resonance imaging may also be used if time permits,^4,12 and especially CT may be helpful in localizing the site of bleeding. ¹⁸ Magnetic resonance imaging is not always available in an emergency setting, but it has the advantage of not using radiation. In our case, CT suggested a large volume hemoperitoneum on the left side of the uterus extending to the upper abdomen, which prompted an urgent laparotomy. There is often concern about the safety of CT imaging for pregnant women. However, the fetal exposure to radiation during CT is estimated to be 5 to 20 mGy, which is below the maximum radiation dose recommended by the American College of Radiology and Society for Pediatric Radiology Guidelines.^19–21 CT imaging is readily available in emergency departments and only takes minutes to obtain in most hospitals in China. Plain CT can approximately determine the location of the bleeding, such as whether it is around the liver, spleen, or in the pelvic area. Plain CT is equally effective in diagnosing hemorrhage to some extent as contrast-enhanced CT and avoids the risk to the fetus from the contrast medium. Plain CT may be considered as a diagnostic tool even in pregnant women when ultrasound is inconclusive and when magnetic resonance imaging is not available. However, plain CT also has its limitations. Plain CT may not accurately determine the cause of bleeding, especially for minor vascular rupture or seepage.

The management of SHiP depends on the patient’s clinical presentation, hemodynamic status, and gestational age. A literature review showed that perinatal and maternal mortality rates for SHiP were 26.9% and 1.7%, respectively. ⁴ Surgical intervention is the primary treatment for SHiP. A midline-longitudinal incision may be helpful to check and confirm areas of bleeding. In the third trimester or late term pregnancy in patients with SHiP, delivering the fetus may be necessary. However, the decision to deliver is more difficult in the early or second trimester. A poor prognosis (fetal death, neonatal death, and miscarriage) of SHiP is more common in patients with less than 32 weeks of gestation. ¹⁶ Extending the gestational age with effective hemostasis by multidisciplinary team evaluation is possible. In our case, because of in vitro fertilization and prematurity, we decided to avoid delivery of the fetus at the first presentation. We asked a surgeon to help us to exclude injury from a liver or a spleen. Fortunately, a subsequent good fetal outcome was obtained, which is a similar finding to that reported by Jayalath et al. ²² Interventional radiology is becoming integral in managing spontaneous hemoperitoneum. Lier et al reported a favorable outcome in a pregnant woman with uterine artery aneurysm rupture and severe anemia treated with uterine artery embolization at 22 weeks’ gestation. ⁴ This finding highlights the potential effectiveness of uterine artery embolization in the management of SHiP.

In conclusion, SHiP is a severe complication of pregnancy with a high risk of emergency surgery, preterm birth, and perinatal death. Endometriosis and artificial reproductive technology are risk factors for SHiP. A differential diagnosis can be challenging because of various causes and non-specific symptoms. There needs to be better awareness of SHiP, and plain CT should be considered when ultrasound is not diagnostic. The collaboration of obstetricians, interventional radiologists, and surgeons using a multidisciplinary approach is crucial for treating SHiP, ensuring individualized care tailored to the specific clinical scenario for optimal outcomes for the mother and the fetus.