Spontaneous hepatic rupture after mitral valve replacement for infective endocarditis in pregnancy: a case report

Abstract

Background

Spontaneous liver rupture during pregnancy is rare and is mostly related to hypertension, eclampsia, or underlying hepatobiliary diseases. Hepatic rupture due to infective endocarditis is even less common.

Case presentation

We report a pregnant woman at 32 weeks gestation without hypertension and underlying hepatobiliary disease who underwent mechanical mitral valve replacement for infective endocarditis. Low molecular weight heparin 0.4 ml q12h was used, and it was changed to warfarin on the third postoperative day (POD). On POD5, the patient developed liver rupture and intrauterine fetal death. After accepting blood transfusion, the hemoglobin level still decreased. On POD6, we performed an exploratory laparotomy and found a rupture in the right lobe of the liver, followed by liver rupture repair packing and cesarean section. But the patient still had obviours abdominal drainage. At night, the interventional radiologist performed an emergency liver arteriography, which revealed bleeding from the right hepatic artery branch, and interventional embolization was performed. Finally, the hemodynamics became stable. The patient was discharged 2 weeks later. Three months after surgery, an enhanced CT scan indicated that the liver had basically returned to normal.

Conclusion

Infection can lead to liver rupture in pregnancy without high blood pressure. Appropriate targeted antibiotic therapy is essential and should be accompanied by prompt surgical or endovascular treatment to improve maternal and fetal outcomes.

Background

Liver rupture occurs in approximately 1 in 200,000 pregnancies, mainly associated with acute fatty liver of pregnancy or HELLP syndrome, and rarely without associated liver disease [1]. To our knowledge, this is the first case report that describes spontaneous liver rupture after cardiac surgery with infective endocarditis in pregnancy.

Case description

A 25-year-old woman at 32 weeks gestation was admitted to the hospital with recurrent fever and left limb weakness that had lasted for more than 2 months. At onset the patient had fever up to 38.2 °C that subsided after an undocumented oral antibiotic course. One week after the fever, the patient suddenly developed left limb weakness and unsteady walking, and she went to the local hospital. The blood culture reported Streptococcus bovis biotype II, and the MRI of the head showed cerebral infarction without specific information. Transthoracic echocardiography (TTE) showed infective endocarditis (IE) of the mitral valve with vegetation and moderate-to-severe regurgitation. The patient refused surgery to preserve the fetus and received empirical vancomycin for an unclear period. Approximately 2 weeks later, the patient’s left limb weakness worsened, accompanied by drowsiness and slurred speech. She went to the local hospital again, and the blood culture was still Streptococcus bovis type II. The MRI of the head showed cerebral infarction with hemorrhage of the right caudate nucleus and lenticular nucleus and multiple chronic cerebral infarctions in the left cerebellar hemisphere, which were not operated on. TTE showed multiple vegetations in the anterior and posterior leaflets of the mitral valve and severe mitral valve regurgitation. After receiving medication and antibiotic treatment for one month, her temperature was normal and neurological symptoms improved. There was no history suggestive of hypertension, eclampsia, or connective tissue disorders. Then she came to our center due to IE.

The patient was conscious, without jaundice and subcutaneous hemorrhage. On physical exam she was afebril, with stable hemodynamics. Her muscle power scale in the left upper limb was MRC 3/5, while it was normal in the left lower limb and right side limb. The blood culture was still Streptococcus bovis type II, sensitive to teicoplanin and clindamycin. The prothrombin time (PT) was 11.3 s and the INR was 1.01. She did not have viral hepatitis, fatty liver, or other underlying liver diseases. Considering the patient was in the late semester of pregnancy, we suggested the patient and her family to undergo a cesarean section before cardiac surgery. However, they were worried about the high cost of intensive care for preterm infants and strongly insisted on undergoing cardiac surgery during pregnancy.

The patient was treated with clindamycin and teicoplanin for one week before median thoracotomy mitral valve replacement. Cardiopulmonary bypass (CPB) was performed with pulsatile perfusion and temperatures maintained between 35.3 °C and 36.6 °C. Numerous vegetations involved both mitral leaflets, with severe leaflet destruction; a #27 Carbomedics mechanical mitral valve was implanted. We continued to monitor the fetal heart rate (FHR) during the operation. The FHR decreased transiently during aortic cross-clamping (ACC), reaching 70 beats/min, before gradually recovering to 150 beats/min after releasing the aortic clamp (Fig. 1A-C). After stopping the CPB, we used the magnesium sulfate 5 g pumped in half an hour to suppress contractions. The CPB time and ACC time were 45 min and 31 min, respectively. There was no obvious abnormality in fetal ultrasound after surgery. The tracheal tube was removed on POD1. The pericardial and mediastinal drainage tubes were removed, and warfarin was started for anticoagulation on POD3.

Fig. 1 .

A-C are fetal heart rate records. The images showing the FHR dropping to 70 beats/min (A) and gradually returning to 140–150 beats/min during surgery (B-C)

On the afternoon of POD5, the patient had a sudden severe abdominal pain, an increased heart rate of 140 beats/min, and a blood pressure of 140/80 mmHg. Then, her urine output decreased, and fetal movement decreased consciously. Arterial blood gas showed hemoglobin 70 g/L (down 40 g/L from the previous day), and lactate of 10 mmol/L. Blood test results revealed elevated liver enzymes [alanine aminotransferase (ALT): 105 U/L, aspartate transaminase (AST): 235 U/L]. Platelets (211 × 10⁹/L) and white blood cell counts (23.71 × 10⁹/L) were highly elevated. The PT rose from 23.5 s on the previous day to 51.9 s, and INR rose from 2.1 to 4.4. We gave the vitamin K1, then the PT and INR decreased to 27.1 s and 2.26, respectively. The patient did not accept the oral warfarin on the day. TTE was normal, but the fetal ultrasound showed the fetus died. On the same day, abdominal CT without contrast demonstrated an intrahepatic hematoma with a small volume of ascites (Fig. 2A-C). After providing 8U leukocyte-reduced red blood cells (LR-RBC) and 400 ml plasma, the patient’s heart rate and blood pressure were stable, but hemoglobin declined to 62 g/L. Therefore, the hepatobiliary surgeon and obstetrician jointly performed an exploratory laparotomy on the 6th postoperative day, using a right incision under the costal margin with a median incorporation. Our multidisciplinary team (MDT) found a subcapsular hematoma of about 8 × 8 cm in the S5 and S6 segments of the right lobe of the liver, and a rupture of 5 cm and 3 cm in length was found on the right edge and upper edge of the hematoma, respectively. After opening the capsule and evacuating the clot, active bleeding was identified from an irregular 3-cm laceration on the right hepatic surface near the gallbladder bed. The liver laceration was repaired with 3 − 0 Prolene^® sutures and hemostatic sponges were applied. Then, there was no evidence of active hemorrhage. A dead fetus was delivered through cesarean section by the obstetrician. Pathological findings indicate liver necrosis with inflammatory cell infiltration (Fig. 3). After surgery, the patient continued to have hypotension, with an intraperitoneal drainage of 500–800 ml/h, and hemoglobin was 44 g/L. Because the conditions did not improve obvioursly after providing 6U LR-RBC and 600 ml plasma transfusion, MDT considered that hepatic bleeding was still present. The interventional doctor performed a hepatic angiography and found extravasation of contrast agent in the distal branch of the right hepatic artery. Finally, 4 pieces of 2 mm×4 cm and 2 pieces of 3 mm×4 cm stainless steel embolization coils (platinum with spaced synthetic fibers, COOK^®) were placed to embolize the culprit artery. After ranscatheter embolization, her hemodynamics were stable, and the intraperitoneal drainage had decreased to 100-150 ml/h. Two weeks after surgery, an abdominal CT showed that most of the intrahepatic hematoma had absorbed (Fig. 2D). Meanwhile, ALT and AST returned to normal values, so the patient was discharged. Three weeks after surgery, the abdominal CT showed the hematoma had shrunk (Fig. 2E). Three months after surgery, an abdominal CT showed the intrahepatic hematoma had completely absorbed (Fig. 2F). During the follow-up period, the patient firmly refused to undergo colonoscopy.

Fig. 2.

A-E are abdominal CT images. Coronal and axial CT without contrast images show (A-C) intrahepatic hematoma with small ascites on POD5; D partial resorption at 2 weeks; E further shrinkage at 5 weeks; F axial contrast-enhanced CT image shows complete resolution at 3 months. Arrows indicate the hematoma

Fig. 3.

The pathological findings indicate liver necrosis with inflammatory cell infiltration (Hematoxylin-eosin staining, ×200)

Discussion

In this case, the patient did not have hypertension or underlying liver disease, but still suffered a liver rupture, which we believe was mostly caused by the rupture of an infectious aneurysm due to IE. IE is present in 0.00002% of delivery hospitalizations [2]. IE-induced peripheral vascular aneurysms are more common in intracranial and splenic arteries [3, 4]. There are also very few cases of intrahepatic aneurysm formation, while streptococcal infections account for 40%, and 90% occur in the right lobe of the liver [5]. Since the onset of the disease, multiple blood cultures revealed bacteremia (lasting nearly three months), and the patient had cerebral infarction before surgery. Therefore infectious aneurysms can occur. We hypothesize that prolonged bacteremia led to arterial wall infection (mycotic aneurysm) or vessel wall weakening, resulting in intrahepatic arterial rupture and subcapsular hematoma. To our knowledge, this is the first case of liver rupture in pregnancy who underwent cardiac surgery because of IE that we have been able to find. Our search terms are ((liver rupture) AND (infective endocarditis)) AND (pregnancy), and we did not find any consistent case report in Pubmed. Postoperative oral anticoagulation with warfarin further exacerbates the spread of intraparenchymal hematoma to form subcapsular hematoma.

Surgery and transcatheter hepatic arterial embolization are both effective treatments. Most previours studies of liver rupture in pregnancy are case reports, Augustin et al., analysed 391 cases of spontaneous hepatic rupture during pregnancy/puerperium complicated by the hypertensive disorder [6]. They found that liver packing did not influence maternal survival, with 164/298 (55.0%) of women who survived and 49/80 (61.3%) of women who died (P = 0.320) [6]. Moreover, 33 women underwent embolization with one death (3.0%) [6]. Liver transplantation has also been an effective salvage in patients with continued decompensation despite standard interventions [7]. 14 women who underwent liver transplantation all survived [6]. We believe that the key to liver rupture is finding the bleeding points and controlling them. In our case, surgery is more suitable for repairing long superficial ruptures, while interventional method is more suitable for embolizing deep vascular ruptures.

The cause of fetal death is more complicated. The fetal mortality of IE during pregnancy could reach 14%, and the most common pathogens were streptococcal (43%) and staphylococcal (26%) species [8]. The fetal mortality rate for spontaneous liver rupture during pregnancy was 37.2% [6]. After comprehensive consideration, we believe that the direct cause of fetal death was fetal ischemia due to maternal liver rupture and massive hemorrhage. The reasons are as follows: (1) The rapid decline in hemoglobin and less fetal movement occurred very closely; (2) there was no abnormality in the fetal ultrasound before liver rupture. Although infection with Streptococcus bovis in pregnant women can cause intrauterine infection and lead to clinical sepsis in neonates [9], the patient had been already treated with targeted antibiotics before surgery. Therefore, the intrauterine infection maybe not the direct reason of fetal death, but its negative impact on the fetus cannot be ignored. It could make the fetus more vulnerable to stimulations and changes in the maternal environment. Therefore, intrauterine infection may be an indirect cause of fetal death. Unfortunately, we were more concerned with emergency liver surgery and did not leave placental or amniotic fluid specimens for bacterial culture. In addition, there was a transient deceleration of the FHR during surgery. But the fetal death directly caused by surgery is more common within 24 h after surgery, so the possibility of fetal death caused by surgery can be basically ruled out.

Reviewing the analysis of this patient, the following experience is noted. (1) For conscious patients who need accept surgeries for IE following stroke, with uncontrolled infection or a high risk of recurrent embolization, surgery is recommended. Cardiac surgery between 13 and 28 weeks gestation is the best time [10]. Therefore, there was maybe a better cardiac surgery timing for this patient at 25 weeks gestation. (2) For the patient at 32 weeks gestation, cesarean section should be considered first, which can not only increase fetal survival but also reduce the impact of volume overload on the heart. In this case, the patient strongly refused to give birth before cardiac surgery because of the high cost of the premature baby. (3) This patient initially presented with abdominal pain and a decrease in hemoglobin, but the blood pressure was normal, so we first chose blood transfusion. When the patient developed a hemodynamic instability, she was diagnosed with liver rupture and underwent emergency surgeries under the assistance of the MDT. Therefore, for pregnant women who do not have hypertension and underlying liver disease, we can’t ignore the possibility of liver rupture, although it is rare.

There are some limitations of this case report. First of all, we did not store the images of embolization to state the exact branch embolized. Secondly, we only did a general pathology of the liver that was not enough to identify the infectious aneurysm. In the end, the ability to determine precise cause of fetal death is limited. Anticoagulation, infection, and maternal liver rupture all have adverse effects on fetal outcomes, but it is difficult to determine which is the primary cause. The intrauterine infection was unclear because of the absence of placental cultures.

Conclusion

Liver rupture is a rare complication of infective endocarditis in pregnancy, with high fetal mortality. It should be suspected in pregnant patients presenting with sudden abdominal pain, elevated liver enzymes and hemodynamic instability even without hypertension; prompt surgical and endovascular management may be lifesaving.

Abbreviations

TTE

Transthoracic echocardiography

IE

Infective endocarditis

CPB

Cardiopulmonary bypass

FHR

Fetal heart rate

ALT

Alanine aminotransferase

AST

Aspartate transaminase

PT

Prothrombin time

APTT

Activated partial thromboplastin time

MDT

Multidisciplinary team

Authors’ contributions

Zhenzhong Wang was responsible for drafting the article. Zhen Zhang was responsible for the implementation of the surgery, collecting data, and postoperative follow-up.

Funding

This work was supported by the Department of Science and Technology of Guangdong Province (No. KTP2020325).

Data availability

The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

Declarations

Ethics approval and consent to participate

The study involving the human participant was reviewed and approved by the Ethics Committee of Guangdong Provincial People’s Hospital (KY-Q-2022-147-01). Informed consent for participation was obtained from the patient involved in the study.

Consent for publication

Informed consent for publication was obtained from the patient involved in the study.

Competing interests

The authors declare no competing interests.