Spontaneous hemoperitoneum in a 29-week pregnancy with a history of endometriosis: A case report and review of the literature

A 30‐year‐old primigravid woman with a spontaneous conception, at 29 weeks of gestation, presented to our obstetrics and gynecology emergency department with complaints of abdominal and back pain. She had sought care at another center with similar complaints 1 week prior, leading to hospitalization. At that time, her cervical length was measured as 22 mm, and she was noted to have polyhydramnios. However, no complications were observed during follow‐up, and she was discharged 3 days later as her symptoms improved. In terms of her medical history, a laparoscopic cystectomy was performed in August 2022 due to a 90 mm × 50 mm endometrioma in the right ovary. However, deep endometriosis and adenomyosis were not observed. She achieved a spontaneous pregnancy 3 months after surgery. At the time of arrival, the patient's vital signs were recorded as follows: pulse 77/min, blood pressure 80/40 mm Hg, temperature 36.2°C, and oxygen saturation of 99%. Obstetric ultrasound revealed a single fetus with a positive heartbeat, increased amniotic fluid volume, and an estimated fetal weight consistent with gestational age. The placenta was visualized as normal on the posterior uterine wall, and the cervical length measured 22 mm. No abundant free fluid or bleeding was observed, ruling out uterine rupture as a concern. The non‐stress test showed reactivity without any contractions. The patient's hemoglobin level at the time of arrival was 8.3 g/dL. Due to intensifying abdominal pain, a general surgery consultation was requested. Thirty minutes later, decelerations appeared on the non‐stress test, and repeat hemoglobin values dropped to 7.2 g/dL, with blood pressure at 70/50 mm Hg and a pulse rate of 95/min. Consequently, the decision was made for laparotomy and emergency delivery of the baby. The procedure was performed under general anesthesia, and entry was made into the abdominal cavity. Upon entering the abdomen, a significant amount of blood and coagulum was observed. Subsequently, the uterus was opened via a Kerr incision, and a male fetus weighing 1555 grams was delivered, with Apgar scores of 4 and 6. Umbilical cord blood gases revealed a pH of 6.8 and an anion gap of −21.4. The newborn was admitted to the Neonatal Intensive Care Unit (NICU). A wide exploration was conducted, involving the removal of a massive coagulum, which amounted to nearly 1000 cc of blood. An active bleeding site, displaying a decidual appearance, was identified on the left side of the uterus near the broad ligament. Bleeding was successfully controlled with sutures. Subsequently, due to observed bleeding in the upper abdomen near the spleen, a general surgeon was summoned to the operation. The current transverse incision was extended superiorly up to the umbilicus along the midline. As no active bleeding site was observed in this region, three drainage tubes were inserted, and the abdomen was closed. The patient received one unit of erythrocyte suspension intraoperatively and two units postoperatively. Additionally, only one unit of fresh frozen plasma (FFP) was infused during the postoperative period. The patient was discharged from the hospital on the fourth day. The newborn was discharged from the hospital on the 48th day with no complications. The patient gave their free informed consent to the anonymous publication of this case. The Bezmialem University Hospital institutional review board (IRB). In committee decided that the publication of this case report did not require its approval.

Shamsi Mehdiyev—Data acquisition, manuscript drafting. Fatma Basak Tanoglu—Data interpretation, manuscript drafting. Esma Demir Altuncu—Data acquisition. Engin Oral— Conception, final approval.

No funding was received for preparing this manuscript.

Spontaneous hemoperitoneum in pregnancy, despite its rarity in medical practice, has been recognized for its poor maternal and fetal outcomes since the 18th century. 8 It continues to pose challenges in terms of prompt diagnosis and management. For this case, we aimed to present the diagnosis and management of a 29‐week pregnant woman with a history of endometriosis surgery. In the literature, there are 67 SHIP cases with endometriosis (Table  1 ). The mean age of the patients was 32.95 ± 4.39, and 68.18% of them were under 35 years of age. In addition, 77.61% of these patients were nulliparous and 14.93% were multiple pregnancies. Those who became pregnant with ART and those who had a history of previous abdominal surgery constituted 41.79% and 52.23% of the patients, respectively. The mean week when SHIP presented was 27.71 ± 7.86, while the week when pregnancy was terminated was calculated as 29.70 ± 7.90. In most cases, the bleeding site was the posterior surface of the uterus. The results together with the patients' initial symptoms, the median amount of bleeding and pregnancy outcomes are shown in Table  2 . Summary of cases with spontaneous hemoperitoneum in pregnancy. Live birth Stillbirth Live birth Live birth Live birth Abbreviation: N/A, not applicable. Demographic and clinic characteristics of patients with spontaneous hemoperitoneum in pregnancy. Note : Data are expressed as mean ± standard deviation, median, minimum, maximum or number (%). Abbreviation: ART, assisted reproductive technology. In a prospective population‐based cohort study conducted by Mazzacco et al. 46 in Italy, it was reported that at the time of hospital admission abdominal pain was present in 89.6% of SHIP patients, hypotensive symptoms in 27.6%, hypovolemic shock in 24.1%, and fetal distress in 37.9%. The differential diagnosis includes conditions involving extra‐pelvic structures such as traumatic splenic artery and vein bleeding, splenic and hepatic injury, and ovarian artery rupture. Additionally, cases involving uterine rupture and placental abruption, which present with abdominal pain and fetal distress, can also lead to massive bleeding. Challenges in early diagnosis arise from symptoms that are non‐specific and can mimic other conditions. A comprehensive review of current literature suggests that early assessment of these patients, manifesting hemodynamic instability or fetal bradycardia, warrants emergency laparotomy without delay for imaging methods. In instances where hemodynamic stability and fetal well‐being are confirmed through imaging modalities such as ultrasound, computed tomography, or magnetic resonance imaging, the application of minimally invasive techniques such as laparoscopy and artery embolization facilitates the continuation of pregnancy and might even prevent complications in newborns resulting from preterm births. 39 , 47 , 48 Given the presence of symptoms such as back and abdominal pain, hypotensive symptoms, and later, the manifestation of fetal distress after initial follow‐up in our patient, a definitive diagnosis was established following explorative laparotomy. However, diagnostic imaging modalities could be used in cases lacking hemodynamic instability and reassuring fetal well‐being. Expectant management could then be considered as an alternative option, aiming to reduce urgent C‐section rates and mitigate negative neonatal outcomes. Although ectopic pregnancy and miscarriages are known to be complications related to endometriosis during pregnancy, SHIP is an acute event associated with maternal and fetal mortality and morbidity that should be kept in mind with a prevalence of 0.4%. 49 When Lier et al., 50 examined the intraoperative biopsies taken from the areas causing bleeding in SHIP cases, they found that the pathological diagnosis was endometriosis in 75% and deciduosis in 25%. Aziz et al. 15 demonstrated medial irregularity, fragility, and widening of vessels lumen and even loss of vascular integrity of vessels in endometriotic lesions. To meet the increasing needs of the placenta and fetus, an increase in plasma volume occurs as pregnancy progresses, and this increase begins to become evident as early as 6–8 weeks and reaches its peak in the 32nd week of pregnancy. 51 Bleeding arises both from increased intraluminal pressure caused by pregnancy and vascular erosions secondary to chronic inflammation prompted by endometriosis. 52 The association of SHIP cases with third trimester can be related to the fact that the anti‐inflammatory effects of progesterone decrease as gestation advances to the third trimester. 44 This process prompts inflammation and peritoneal hemorrhages. Similar to our case, a review of cases by Mazocca et al. 46 reveals that 65% of SHIP cases had previously undergone abdominal surgery, which increases intrabdominal inflammation secondary to previous surgery, fragility of vessel walls, and increased peritoneal tension due to abdominal adhesions predisposed to bleeding. The maternal mortality rate, which was 49% in the review conducted by Hodgkinson et al., 53 encompassing 75 cases in 1950, has significantly declined to 0%–0.4% due to developments in advanced life support methods, modernization in anesthesia, and improved surgical techniques. However, perinatal mortality rates remain persistently high, ranging from 31% to 36%. By successfully navigating diagnostic pathways, expectant management can be considered as an alternative to emergency C‐section. Additionally, the use of embolization and the availability and feasibility of laparoscopic approaches might contribute to reducing preterm birth rates and mitigating negative neonatal outcomes by allowing the continuation of pregnancy. In conclusion, it is crucial to consider SHIP in pregnancy, especially in pregnant patients with a history of endometriosis surgery. Managing such high‐risk cases in specialized centers and easily identifying predisposing factors for SHIP can lead to improved outcomes, despite its rarity and poor prognosis.

Endometriosis is a common gynecological disease that affects at least 10% of women in reproductive age. 1 Although it is known as one of the causes of infertility, it is also associated with negative pregnancy outcomes. Miscarriage, preterm birth, placenta‐associated complications (pre‐eclampsia, abruptio placenta, and placenta previa), hemorrhage, acute abdomen (spontaneous uro‐hemoperitoneum, uterine rupture, and bowel perforation), fetal dystocia, stillbirth, and cesarean delivery are among these negative outcomes . 2 Endometriosis was shown to be present in 71% of spontaneous hemoperitoneum in pregnancy (SHIP) cases. 3 SHIP is defined as sudden, nontraumatic intraperitoneal bleeding that occurs during pregnancy or up to 42 days postpartum. 4 The incidence ranges between 4 and 4.9 per 100 000 births. 5 Although seen rarely, it is associated with perinatal morbidity and mortality due to maternal hemodynamic instability. Pregnancies in women older than 35, previous history of abdominal surgery, use of assisted reproductive technology (ART) for conception, multiple pregnancy, and endometriosis are some of the known risk factors for SHIP. 6 , 7 SHIP is most commonly encountered during the second half of pregnancy and necessitates emergency surgical intervention. This often results in high neonatal mortality rates secondary to preterm birth. Due to its nonspecific clinical presentation, including symptoms such as abdominal pain, signs of hypovolemic shock, and fetal distress, the final diagnosis is typically established intraoperatively. Despite its rare occurrence, early diagnosis and appropriate management are of utmost significance, given its potential to be a life‐threatening condition. In this study, we present the case of a 30‐year‐old 29‐week pregnant patient with a history of endometriosis surgery who was admitted to our emergency department with SHIP.

The authors declare they have nothing to disclose.