Multiple Episodes of Spontaneous Hemoperitoneum in Pregnancy: A Case Report on Diagnosis and Management

Introduction Spontaneous hemoperitoneum in pregnancy (SHiP) is a rare but potentially life-threatening condition, associated with significant maternal and fetal morbidity and mortality. SHiP is characterized by sudden and nontraumatic intraperitoneal bleeding during pregnancy and up to 42 days postpartum. Case presentation A 34-year-old Gravida 2, Para 1, Caucasian Belgian woman presented at the emergency department of a large regional hospital at 29 weeks and 3 days with abdominal pain. After investigation, the diagnosis of SHiP was made and treated conservatively because of spontaneous resolution of complaints. Two weeks after initial presentation she was readmitted with the same complaint; however, this time she was hemodynamic unstable due to the large hematoperitoneum requiring an emergency cesarean section. Her postpartum care was complicated by a third episode day 1 postpartum due to a pseudoaneurysm.

Management of SHiP is reliant on three pillars: prompt recognition and diagnosis, timely and correct intervention, and adequate follow-up care. Awareness on SHiP risk factors and diagnosis is growing due to more comprehensive studies, systematic reviews, and more appreciation of the risks of pregnancy after endometriosis surgery or endometriosis in situ. However, diagnostic delay due to misdiagnosis remains a significant issue in SHiP cases. Management of SHiP encompasses timely advanced life support, treatment of hypovolemic shock if present, surgical treatment, and in selected cases conservative treatment could be justified. The risks and benefits of conservative and ambulatory management of SHiP should be further investigated.

Symptoms of SHiP are nonspecific. Early diagnosis is of importance since this clinical entity can be life threatening and requires intervention in most cases. In selected cases, conservative management can be an option, but further research is needed to identify which clinical and imaging variables are important for this strategy, and threshold for surgical intervention and even delivery should be low. 1. Introduction Spontaneous hemoperitoneum in pregnancy (SHiP) is a rare but potentially life-threatening condition, associated with significant maternal and fetal morbidity and mortality [1, 2]. SHiP is characterized by sudden and nontraumatic intraperitoneal bleeding during pregnancy and up to 42 days postpartum [1]. Due to its rarity and often underreported nature, the incidence of SHiP is hard to determine but has been estimated at four in 100,000 live births [1–3]. Diagnosing this obstetric emergency is challenging because of its nonspecific presentation, which can include subacute abdominal or flank pain, hypovolemic shock, and fetal distress [1–13]. The etiologies of SHiP are diverse and include ruptured utero-ovarian vessels, spontaneous rupture of the uterus or other abdominal organs but endometriosis, and certainly decidualized endometriosis, is the most common observed underlying condition [13, 14]. Risk factors include endometriosis, the use of artificial reproductive techniques, advanced maternal age (over 35 years), and multiple pregnancies [1–3, 13]. This case report aims to illustrate the diversity of the clinical symptoms of SHiP, its possible causes, complications, and treatment to facilitate future identification and management of this condition. Prompt recognition and treatment are crucial for obstetricians, emergency physicians, and all healthcare providers involved in fetomaternal care. 2. Case Presentation A 34-year-old Gravida 2, Para 1, Caucasian Belgian woman presented at the emergency department of a large regional hospital at the gestational age of 29 weeks and 3 days. She presented with acute abdominal pain, after making a wrong movement getting out of her car, rated 10 on the Numerical Rating Scale (NRS). She did not report any vaginal bleeding, nor any fluid loss, contractions, gastrointestinal, urinary, or other general complaints. The patient had no significant medical history other than a benign osteoma of the forehead 10 years ago and vitiligo of the hands for the past 8 years. She has conceived easily in both pregnancies. Clinical examination showed a non-tender abdomen, with nonspecific sensitivity to deep palpation. Clinical vital parameters were all normal with a blood pressure of 113/62 mmHg, heart rate of 65 beats per minute, and temperature of 36.3°C. CTG was reactive and normal for gestational age. Obstetrical ultrasound showed a normal fetal growth and biophysical profile. Cervical length was estimated at 21 mm, through transvaginal ultrasound. Laboratory results at that time were normal: hemoglobin (Hb) = 11.7 g/dL (reference range: 11.7–15.1 g/dL), white blood cell (WBC) count = 12.2 × 109/L (reference range: 4.3–9.6 × 109/L), and C-reactive protein (CRP) = 2.8 mg/L (which increased to 17 mg/L on day 1 postadmission). Kidney and liver function tests were within normal range, as were prothrombin time (PT), international normalized ratio (INR), and activated partial thromboplastin time (aPTT). Further investigations to exclude appendicitis and pyelonephritis were initiated. An abdominal ultrasound reported a limited amount of fluid in the perisplenic, perihepatic, and Douglas pouch regions, preferably physiological. The appendix was not visualized, and the gallbladder and kidneys appeared normal. At that time, the presumed diagnosis was appendicitis for which a course of antibiotics was started. Fetal maturation was also initiated as pain was severe, and maternal cause for preterm need for delivery could not be excluded. A second look ultrasound was performed by a senior radiologist on day 1 postadmission, which described a noticeable increase in free abdominal fluid compared to original imaging prompting an emergent MRI, which was performed 19 h postadmission. Free abdominal fluid was confirmed in both the right flank area and the pouch of Douglas, most consistent with pus of an unknown origin. Neither the appendix nor endometriosis were visualized. There was also no active bleed. After multidisciplinary discussion between the senior gynecologist and senior radiologist, it was agreed that these findings could also be compatible with blood instead of pus, thus leaving SHiP as the most probable diagnosis. The diagnosis of SHiP was made 25 h postadmission. At the timing of diagnosis, the multidisciplinary team concluded that a conservative approach was defendable due to the absence of active bleeding on MRI, resolution of symptoms at the time, and stable hemodynamics in combination with a very preterm gestational age. This decision was made after careful consideration and daily reassessment. She could be discharged on day 5 postadmission in relatively good general health. A teleconsult 5 days after discharge was reassuring. Exactly 9 days after discharge, at a gestational age of 31 weeks and 2 days, she presented again with acute abdominal pain in the right iliac fossa (NRS 10) slightly worse than the previous time. She did not report any vaginal bleeding, nor any fluid loss, contractions, gastrointestinal, urinary, or other general complaints once again. She was clinically stable with vital parameters as follows: blood pressure 108/74 mmHg, heart rate 68 beats per minute, afebrile, and oxygen saturation of 99% on room air. Abdominal examination revealed a tender abdomen without a specific focal point. A preliminary obstetric ultrasound showed a good fetal heart rate and no signs of uterine rupture or placental abruption. She was immediately transferred to the maternal intensive care unit for further investigation. During the second episode, further investigations showed a Hb of 10.7 g/dL with no other abnormalities. Abdominal ultrasound by a radiologist and gynecologist revealed fluid in the perihepatic, perisplenic, and both flank regions, with at least two pockets exceeding 30 mm. Vaginal ultrasound showed a closed cervix measuring 26 mm and fluid in the Douglas pouch (Figure 1). Considering the previous episode and the likely diagnosis of SHiP, the fluid was presumed to be blood. Despite attempts, the patient deteriorated and became hemodynamic instable. An urgent cesarean section was performed 3 h postadmission. Fetal heart rate remained optimal throughout the whole episode and her baby was born at 31 weeks and 3 days gestational age, with Apgar scores of 7, 9, and 9, and a weight of 1700 g. Intraoperatively, the presumed diagnosis of SHiP was confirmed, with an estimated blood loss of 1800 mL (300 mL attributed to the cesarean section and 1500 mL due to the hemoperitoneum). A lesion was identified on the posterior wall of the right broad ligament (Figure 2), histological examination confirmed decidualized cells compatible with endometriosis. The abdomen was inspected for additional lesions with a general surgeon, none were found. The patient received one unit of packed red blood cells immediately following cesarean. Two abdominal drains were placed, which subsequently collected 180 and 20 mL of bloody fluid 4 h postpartum, attributed to further dissolution of blood clots. Twelve hours postcesarean section, the patient rapidly deteriorated and was transferred to the emergency room due to hemodynamic instability. Hb levels dropped from 10.9 to 8.0 g/dL even though she had received 1 unit of packed cells intraoperatively. An emergency computed tomography (CT) scan revealed an active bleed from a large pseudoaneurysm of an internal iliac subartery, most likely the uterine artery (Figure 3). The pseudoaneurysm was successfully embolized using coil embolization. The patient was transferred to the intensive care unit, where she received an additional two units of packed red blood cells. On day 2 postcesarean and day 1 postcoiling, the patient returned to the maternal intensive care ward where the first drain was removed. The second drain was removed on day 7 postcesarean. On day 4 postcesarean, she experienced another episode of acute abdominal pain; however, no active bleeding was discovered on CT, and pain quickly resolved. There were no further complications, and the patient was discharged on day 11 postcesarean. The infant remained hospitalized due to prematurity, but no significant complications occurred during the hospital stay. Three months later she had a postpartum follow-up with an experienced sonographer showing a residual hematoma of 21 mm × 15 mm × 18 mm cranial of the right uterine horn as well as an avascular solid mass of 40 mm × 11 mm × 13 mm adherent to the right posterior uterine wall, most compatible with another hematoma (Figure 4). No signs of deep invading endometriosis were found. Another 2 months later (postpartum month 5), an ultrasound showed resorption of the previous described hematoma on the right sight and visualized an endometriotic nodule invading the parametrium. Follow-up magnetic resonance imaging was inconclusive describing a residual hematoma in the right paraovarium parametrium. An underlying deep infiltrating endometriotic lesion could not be excluded. As complaints had resolved at a teleconsult 6 months postpartum a conservative approach was proposed. A follow-up appointment in 6 months was scheduled. An overview of this timeline of events can be found in Table 1. | Event | Timing | Actions | |---|---|---| | Initial presentation | 29 weeks 3 days AD | - • Intake, first investigations (parameters, labs, abdominal, and vaginal ultrasound) and stabilization - • Presumed diagnosis: appendicitis. - • Antibiotics started - • Fetal maturation started due to possible risk of preterm delivery | | Investigations and discharge | 29 weeks 4 days AD | - • A second ultrasound showed an increase in free abdominal fluid compared to initial imaging - • MRI performed 19 h after admission - • Multidisciplinary discussion: most likely diagnosis of SHiP - • Antibiotics and corticosteroids were discontinued after 3 doses of corticosteroids - • The patient remained stable and was discharged on day 5 postadmission in good general health | | Second presentation | 31 weeks 2 days AD | - • 9 days later: patient presented again with acute abdominal pain (NRS 10), slightly worse than the previous episode - • Intake, first investigations (parameters, labs, abdominal, and vaginal ultrasound) and stabilization - • Diagnosis of second episode of SHiP | | Emergency cesarean section | 31 weeks 3 days AD (3 h postadmission) | - Developing hemodynamic instability - • An urgent cesarean section was performed - • Baby born at gestational age 31 weeks 3 days, with Apgar scores 7, 9, and 9, weighing 1700 g - • Estimated blood loss: 1800 mL (300 mL due to cesarean, 1500 mL due to hemoperitoneum) - • A lesion on the posterior wall of the right broad ligament was identified and confirmed as decidualized endometriosis through histological examination - • The patient received 1 unit of packed red blood cells post-surgery - • Two abdominal drains were placed | | Hemodynamic instability and emergency CT scan | 12 h postpartum | - Hemodynamic instability - • An emergency CT scan revealed a large pseudoaneurysm of the uterine artery - • Coil embolization of the pseudoaneurysm - • Additional 2 units of packed red blood cells - • Transfer to intensive care | | Postcoiling recovery | Day 2–11 postpartum | - • Transfer maternal intensive care - • First abdominal drain was removed on day 2 - • Negative CT scan on day 4 after episode of acute pain - • Second drain was removed on day 7 postcesarean. | | Discharge | Day 11 postpartum | • Discharge in good clinical health | | Postpartum follow-up 1 | Month 3 postpartum | - • Ultrasound showed a residual hematoma (21 mm × 15 mm × 18 mm) cranial to the right uterine horn and a solid mass (40 mm ×11 mm × 13 mm) adherent to the right posterior uterine wall, suggestive of another hematoma - • No signs of deep endometriosis were found | | Postpartum follow-up 2 | Month 5 postpartum | - • The hematoma on the right side showed signs of resorption, raising suspicion of an endometriotic nodule invading the parametrium - • Follow-up MRI was inconclusive, but a residual hematoma was noted in the right paraovarian parametrium, with suspicion of deep infiltrating endometriosis | | Postpartum follow-up 3 | Month 6 postpartum | - • Teleconsult - • Complaints resolved: conservative approach - • Follow-up appointment scheduled in 6 months | 3. Discussion This case report describes a patient who experienced several episodes of SHiP and how it was managed. We hope it will add to the slowly increasing awareness of this obstetric emergency. Management of SHiP is reliant on three pillars: prompt recognition and diagnosis, timely and correct intervention, and follow-up care. 3.1. Diagnosis Presentation and risk factors for SHiP are at this time better known due to more comprehensive studies and systematic reviews [1–3]. This patient, however, did not exhibit any risk factor for SHiP, such as endometriosis, assisted reproductive technologies (ARTs), or advanced maternal age (>35 years) [1–3]. However, it has been described that endometriosis is not diagnosed until SHiP occurs during pregnancy in approximately33% of cases [1]. Our case underscores the need for increased awareness of SHiP even in individuals without risk factors as this caused diagnostic delay in our case in which the patient first presented during on-call hours. The symptoms presented by this patient were typical, most notably acute abdominal pain, which is the most frequent symptom [1–13]. Her pain, however, was more pronounced on the right lateral side, whereas the left side is more commonly reported [1]. Nonetheless, acute abdominal pain in pregnancy should always prompt consideration of SHiP as a differential diagnosis, especially with simultaneous abdominal fluid [1–12]. Diagnostic delay remains a significant issue in SHiP cases, even when presentation is typical [1, 6]. This patient presented during on-call hours both times, limiting diagnostic possibilities. The second time, there was no diagnostic delay due to the prior episode serving as a reference and the more urgent setting of this episode. Only an ultrasound was performed, which is still the most used diagnostic tool [1–4, 11]. The initial episode, however, took 24 h to diagnose, as the patient underwent two ultrasounds followed by an MRI, which first suggested an infectious process, but later confirmed the presence of blood. This is consistent with reported delays ranging from 2 h to 5 days [1, 6]. In this case, the underlying cause of SHiP was decidualized endometriotic tissue, which appears to be a more pronounced risk factor than nondecidualized endometriosis [14]. During pregnancy, a link between ectopic decidualization, particularly that occurring in endometriotic foci, and occurrence of SHiP has been described [13, 14], for the pathophysiology behind SHiP we refer to the article by Rafi et al. [13]. In addition, subclinical bleeding of decidualized tissue may be a potential risk factor for preterm labor [14]. 3.2. Intervention Management of SHiP encompasses timely advanced life support, treatment of hypovolemic shock if present, surgical intervention, and in selected cases conservative treatment [1, 3, 7]. Conservative management strategies have been described and proposed in specific cases by Lier et al. [1]. Two case reports describe conservative treatment in three cases [15, 16]. One patient did not require surgical intervention as fetal demise had been diagnosed, she was resuscitated and after stabilization induced with syntocinon. In the postpartum period, she was diagnosed with endometriosis stage III–IV (rASRM) [15]. The second case was treated with laparotomic intervention a week prior for the first event, and patient was stable enough not to require reintervention during the second event [16]. The third case occurred during labor, with a second episode being treated 30 days postpartum through Pfannenstiel laparotomy after conversion from laparoscopic intervention [16]. Both patients were known with endometriosis stage III–IV (rASRM) [16]. During the first episode of our case, a conservative approach was successfully employed, extending the gestational age by 2 weeks. This renders our case unique as, to our knowledge, it is the first to have been conservatively managed during a first episode of SHiP while not in active labor. This makes the management of ambulatory care experimental, and adequate information should be given to the patient, given that recurrence of SHiP is estimated to occur in approximately 10% of cases [1]. Moreover, there is insufficient knowledge about the risks to the fetus (and mother) in this obstetric emergency, even when seemingly adequate control is achieved. Conservative and ambulatory management of SHiP should be further investigated, as fetal morbidity remains significantly higher than maternal morbidity [1, 5, 11, 15]. Additionally, conservative care should only be considered after multidisciplinary discussions in which all involved healthcare workers feel that findings, taking gestational age into consideration, can warrant this approach. 3.3. Postpartum Care The third episode may be considered traumatic rather than spontaneous, though it is unclear whether the pseudoaneurysm already preexisted due to deeper unnoticed endometriotic lesions or was extravagated by cesarean section. Both explanations have been priorly described as plausible causes for pseudoaneurysms [17–19]. However, this third episode further highlights the importance of a multidisciplinary approach and the necessity of increased awareness of both SHiP and other causes for deterioration such as spontaneous aneurysm hemorrhages [17]. Moreover, it also emphasizes the need for quality postpartum follow-up care, as episodes can occur up to 42 days postpartum [1, 16]. Lastly, as shown in our case, follow-up care is also important to diagnose residual lesions, ensuring qualitative and individualized long-term care for each patient; however, no guidelines on follow-up strategies of patients who experienced SHiP have been found. 4. Conclusion SHiP is a rare and serious obstetric emergency. While it is mostly associated with decidualization and endometriosis, clinicians should always consider SHiP even in patients with no known history of endometriosis. Symptoms of SHiP are nonspecific, and SHiP remains easily missed. The importance of a multidisciplinary approach to address the complexities of SHiP and to improve maternal and fetal outcomes is emphasized. In selected cases, conservative management can be an option, but further research is needed to identify which clinical and imaging variables are important for this strategy, and threshold for surgical intervention and even delivery should be low, given the high rates of fetal (and maternal) demise. In conclusion, ongoing research and the accumulation of case reports, series, and cohort studies are necessary to enhance the understanding and management of SHiP. This will ultimately improve both maternal and fetal well-being. Acknowledgments We would like to thank all personnel involved in the care of our patient. Especially for their expertise in this case, but also for their consistent and qualitative care of all patients in our obstetrics and gynecology department. Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Ethics Statement All guidelines of the Declaration of Helsinki—Ethical Principles for Medical Research Involving Human Subjects were respected. Qualitative care for our patient was our first and foremost priority. Informed consent was obtained. Consent Oral informed consent was provided by the patient. Conflicts of Interest The authors declare no conflicts of interest. Data Availability Statement Data sharing is not applicable to this article as no new data were created or analyzed in this study.