A 37-year-old G2P1 woman of Indian origin was admitted in the Obstetrical Department of the University Hospital of Verona with a 32-week dichorionic twin gestation. She complained of moderate dyspnea (SpO2 98%) and sudden ankle swelling; her blood pressure was high and had significant proteinuria (>30 mg/mmol on spot protein-creatinine). Obstetric history included a term vaginal delivery and a laparoscopic salpingectomy for ectopic pregnancy. She had conceived the index pregnancy after an in vitro fertilization and embryo transfer (IVF-ET) in India. In her country of origin, she had gone through regular antenatal visits, receiving vaginal progesterone, anticoagulant therapy with LMWH for multiple venous thromboembolism risk factors, and levothyroxine due to a pregestational autoimmune hypothyroidism. Prophylactic cerclage had also been placed in India at 14 weeks where she was started on a daily low-dose aspirin regimen.
At admission, treatment was started with oral labetalol 100 mg every 8 hours and a 2-day betamethasone course for fetal lung maturation. Aspirin was discontinued, while LMWH was kept, due to multiple risk factors: age (37 years), twin pregnancy, IVF/ART, and preeclampsia. Twenty-four-hour proteinuria was 2,1 g/d and preeclampsia was confirmed. Platelet count showed a reduction from 125 x 109/L at admission to 98 x 109/L. Ultrasound evaluation showed a normal growth, normal amniotic fluid, and Doppler indices of uterine blood flow for both fetuses.
At 33 weeks of gestation, the patient was delivered with a C-section under general anesthesia due to an uncontrolled rise of the blood pressure failing to respond to treatment (160/100 mmHg) and a further platelet count reduction (91,000/dL). Bleeding amounted to 600 mL. After C-section, no evidence of coagulopathy was observed with stable platelet count. Hemoglobin levels (11.5 g/dL) as well as other coagulation indexes remained in the normal range. The newborns weighted 2090 g and 1830 g and were transferred to the Neonatology Intensive Care unit to be treated for prematurity.
After Cesarean, a sudden increase of blood pressure (175/110 mmHg) was observed and intravenous labetalol was started along with magnesium sulphate for eclampsia prevention. LMWH was continued to prevent postpartum and postoperative DVT. Treatment effectively lowered the blood pressure and obtained a stable condition, with the patient only complaining of moderate heartburn and occasional vomiting. Accordingly, H2 receptor antagonists therapy was started. On the first postoperative day, the patient suddenly complained of a right side retrosternal pain extending to the shoulder blade, nausea, vomiting, and dysphagia. Occurrence of HELLP syndrome was ruled out since platelet count and liver enzymes were normal and no signs of hemolysis were recorded. ECG and chest X-ray were both negative. The following day hematemesis was observed which warranted the indication of a gastroscopy. In the initial phase of the endoscopic evaluation, an eccentric mass was seen, completely obliterating the lumen of the esophagus, which prompted the end of the procedure (Figure 1).
A chest CT scan showed bilateral pleural effusion and confirmed an intramural hematoma of the posterior esophageal wall, extending 15 cm caudally (Figure 2); no active bleeding was seen.
The patient was transferred to ICU where she remained hemodynamically stable, with no sign of perforation and no mediastinal involvement. A conservative management was chosen: supportive care and parenteral nutrition, administration of proton pump inhibitors to decrease gastric acid production, and broad-spectrum antibiotic therapy.
The chest CT control two days later showed no modification of the hematoma with no active bleeding and improvement of the pleural effusion.
Nine days after the initial diagnosis and eleven days after C-section, a new episode of hematemesis required a repeat CT scan. This time a reduction of the hematoma was seen with concomitant blood collection in the stomach. Gastroscopy was indicated, blood content aspirated, and the active bleeding controlled with local therapy. The patient was transfused with 2 units of packed red cells.
After this episode, the patient was discharged from ICU and transferred to a surgical ward where conservative treatment was continued until restarting of food intake; the subsequent course was uncomplicated.
A control chest CT, 17 days after C-section, showed an open lumen, a reduction of the thickness of the esophageal wall, and an almost complete resolution of the parietal hematoma (Figure 3).
The patient was discharged asymptomatic 20 days after the initial diagnosis under antihypertensive and antacid therapy. A detailed chart including medications, laboratories results, images, and procedures throughout hospitalization is presented in Table 1.