Spontaneous Rupture of the Internal Thoracic Artery Causing a Mediastinal Hematoma in a Patient with Alcoholic Liver Cirrhosis: A Case Report

Abstract

Spontaneous arterial bleeding is uncommon, and a mediastinal hematoma caused by spontaneous rupture of the internal thoracic artery has not been reported previously. Patients with liver cirrhosis or heavy alcohol consumption have a higher risk of hemorrhage than those without cirrhosis or excessive alcohol consumption. We present the case of a 39-year-old female with a history of alcoholic liver cirrhosis, who presented with a large mediastinal hematoma attributable to spontaneous rupture of the internal thoracic artery.

INTRODUCTION

Spontaneous arterial bleeding is uncommon, and patients with liver cirrhosis or heavy alcohol consumption have an increased risk of hemorrhage (1). There have been a few case reports of spontaneous rupture of arteries, mainly in alcoholic liver cirrhosis patients (2,3). To the best of our knowledge, a mediastinal hematoma caused by a spontaneous internal thoracic artery rupture has not been reported. Here, we describe the case of a 39-year-old female with known alcoholic liver cirrhosis without a history of trauma or bleeding disorder who presented with a huge mediastinal hematoma considered to be due to spontaneous internal thoracic artery rupture.

CASE REPORT

A 39-year-old female with alcoholic liver cirrhosis was admitted for a 2-week history of jaundice. Physical examination revealed icteric sclerae and skin with abdominal distension. The patient’s vital signs were normal. She had been a heavy drinker for 30 years, consuming at least 140 g of alcohol daily. Initial chest radiography showed no significant abnormalities. In laboratory tests, her hemoglobin (Hb) was 7.8 g/dL (normal range: 11–15 g/dL), total bilirubin was 7.1 mg/dL (normal range: 0.2–1.2 mg/dL), prothrombin time-international normalized ratio (PT-INR) was 2.13 (normal range: 0.85–1.2), and serum albumin was 2.3 g/dL (normal range: 3.5–5.2 g/dL). The Child-Pugh score was C11, and the model for end-stage liver disease (MELD) score was 25.

The patient was clinically suspected of having acute hepatopathy with underlying alcoholic liver cirrhosis. Thus, she received conservative medical management with blood transfusion to correct liver function and US-guided drainage catheter insertion for ascites.

On the 12th day after admission, the patient experienced shortness of breath and chest discomfort. Chest radiography revealed left pleural effusion with newly noted mediastinal widening (Fig. 1A). A drainage catheter was inserted in the left pleural cavity under US guidance, and bloody fluid was drained. To identify the bleeding focus, contrast-enhanced chest and abdominal CT were performed after 2 days, which revealed an 8.7 cm × 6.6 cm × 9.0 cm sized hematoma in the anterior mediastinum at the lateral aspect of the aortic arch with a linear enhancing lesion inside it, which was suspected to be active extravasation (Fig. 1B). The lesion was hyperdense on a nonenhanced scan, with a measured Hounsfield unit of 61, and the enhancement was not clear (Fig. 1B).

Fig. 1. A 39-year-old female with alcoholic liver cirrhosis who presented with 2 weeks of jaundice.

A. Chest radiograph obtained on the 12th post-admission day shows massive left-sided pleural effusion.

B. Contrast-enhanced chest CT scan obtained 4 days later shows a hematoma (8.7 cm × 6.6 cm × 9.0 cm) in the anterior mediastinum at the lateral aspect of the aortic arch. Contrast-enhanced images (upper images) showing internal linear vessel-like enhancing lesions (arrows), suggestive of active extravasation. The lesions appear hyperdense (61 Hounsfield units) on a non-enhanced scan (lower middle image). Enhancement is unclear in these images. Heterogeneous hepatic enhancement with hepatosplenomegaly is observed on an upper abdominal scan (lower left image); these findings are consistent with hepatopathy.

C. Selective arteriography scan obtained the same day showing extravasation in the territory of the left anterior intercostal artery (arrow, left image). Image shows embolization of the left internal thoracic artery using Nester and Interlock coils (arrows, right image).

D. Follow-up CT angiography scan obtained 4 days after embolization show an increase in the size of the mediastinal hematoma, which extends from the thoracic inlet to the cardiac border on a coronal scan (left image). Scans obtained in both arterial (middle image) and venous phases (right image) showing linear enhancing lesions (arrows), which were considered to represent active extravasation.

E. Selective arteriography scan obtained the same day showing extravasation at the upper portion of the left internal thoracic artery, proximal to the site of previous coil embolization (arrow, left image). Embolization of the left internal thoracic artery was performed using a 1:4 glue-lipiodol mixture. Intraoperatively, the distal microcatheter was cast together with glue materials. Image showing the stent deployed into the left subclavian artery to prevent reflux of the catheter (right image).

F. Follow-up chest radiograph obtained 2 months postoperatively showing near-complete resolution of the mediastinal hematoma and pleural effusion.

Selective arteriography was performed immediately after CT for accurate diagnosis, and extravasation of contrast media was observed at the left anterior intercostal arteries (Fig. 1C). Subsequent transcatheter arterial embolization (TAE) was performed and embolization of the left internal thoracic artery was performed using microcoils (Nester^®; Cook, Bloomington, IN, USA) and interlocking detachable coils (Interlock^®; Boston Scientific, Natick, MA, USA) (Fig. 1C). After checking for no further extravasation on control arteriography, the procedure was terminated.

Despite embolization, a daily follow-up chest radiograph after the procedure showed an increasing amount of pleural effusion and an increased size of the mediastinal hematoma. The patient’s Hb level dropped to 6.3 g/dL two days after embolization. Suspecting persistent bleeding, CT angiography was performed to identify another bleeding focus 4 days after embolization. CT angiography showed increased hematoma size (9.0 cm × 6.6 cm × 11.0 cm) with internal suspicious extravasations (Fig. 1D). The amount of left pleural effusion had also increased. Selective arteriography was performed on the same day to evaluate active bleeding, and extravasation was discovered in the left internal thoracic artery just proximal to the previous coil embolization site (Fig. 1E). Embolization of the left internal thoracic artery was performed using a glue (N-butyl cyanoacrylate; Histoacryl, B. Braun, Melsungen, Germany)-Lipiodol (Guerbet, Aulnay-sous-Bois, France) 1:4 mixture (Fig. 1E). Intraoperatively, the distal portion of the microcatheter was cast together with glue. A subsequent stent (Epic 8.0 mm × 40 mm; Boston Scientific) deployment was performed on the left subclavian artery to prevent reflux of the distal portion of the cast catheter (Fig. 1E).

Subsequently, the amount of bloody fluid drained through the chest catheter gradually decreased, and the patient’s symptoms improved daily. Twenty-eight days after the second TAE, the chest catheter was removed because of resolved pleural effusion. The patient’s Hb level increased from 8.3 g/dL, just after the second TAE, to 9.5 g/dL when the catheter was removed. Although other laboratory tests showed no significant improvement (bilirubin, 7.6 mg/dL; PT-INR, 2.19; and serum albumin, 3.8 g/dL), the subsequent clinical course was favorable and the patient was discharged.

She visited the hospital several times for outpatient care for regular follow-up, and chest radiographs showed that the size of the mediastinal hematoma had slowly decreased. Approximately 2 months after discharge, chest radiography showed an almost completely resolved state of previous mediastinal hematoma and pleural effusion (Fig. 1F).

The patient underwent liver transplantation 8 months later, and there was no recurrent bleeding.

This case report was exempt from the ethical approval in our institution. This study was performed according to the latest ethical principles in the Declaration of Helsinki (2013).

DISCUSSION

Patients with hepatic cirrhosis frequently experience changes in hemostasis (4). These alterations are caused by thrombocytopenia, altered platelet function, low levels of the majority of procoagulants and certain anticoagulants, and low amounts of the fibrinolytic protein complex (5). It was previously thought that people with liver cirrhosis had auto-anticoagulation. However, the perception of this conventional idea of hemostasis in patients with liver cirrhosis has been altered. Clinical and laboratory data show that the simultaneous lowering of procoagulant and anticoagulant factors leads to rebalancing of the hemostatic system (5). However, the hemostatic balance in patients with cirrhosis is less stable than that in healthy people, and several conditions, including portal hypertension, endothelial dysfunction, bacterial infection, and renal failure, cause decrease the hemostatic coagulability (1,5).

The common clinical manifestation of hepatic coagulopathy is gastrointestinal hemorrhage. Significant bleeding in advanced liver disease is usually caused by gastroesophageal varices or peptic ulcers, and approximately 30% of liver cirrhosis patients with gastroesophageal varices experience gastrointestinal bleeding in their lifetime (6). Other non-gastrointestinal bleeding causes include more common presentations, such as easy bruising and gum, nose bleeding, and spontaneous arterial bleeding. Spontaneous arterial bleeding is a rare complication of alcoholic liver cirrhosis, and most studies have reported the presence of muscular branching arteries that result in muscle hematoma. The iliopsoas and rectus muscles are common sites for muscle hematoma, which usually occurs after significant trauma (3). However, in patients with significant hepatic coagulopathy, even minor trauma, such as a cough-induced rise in abdominal pressure, can cause hematoma in the abdominal muscles (3).

Acute mediastinal hemorrhage has various causes, such as thoracic trauma, mediastinal great vessel aneurysm, dissection or rupture, or iatrogenic causes, including invasive procedures (7). However, spontaneous atraumatic mediastinal hematoma is rare, with most previously reported causes as follows: complications of enlarged mediastinal masses, hypertension, and alterations in hemostasis (7). The most common clinical presentation of spontaneous mediastinal hematoma is dyspnea, and other presenting symptoms include chest or neck ecchymosis, dysphagia, chest pain, and tachycardia (7). The gold standard diagnostic modalities are angiography and surgical exploration, and most previously reported cases showed no definite bleeding vascular focus on CT or MR images only, especially for spontaneous mediastinal hematoma associated with hemostatic abnormalities.

Our patient did not have any symptoms or color change on the chest wall, although CT showed an active arterial bleeding lesion on the contrast enhancement scan. She had alcoholic liver cirrhosis and altered hemostasis, which may produce a high bleeding tendency but no clinical history of a traumatic event or iatrogenic vascular procedure that could produce rupture of the internal thoracic artery. In previously reported cases of spontaneous arterial bleeding in liver cirrhosis, the majority of bleeding arteries were muscular branches, such as the intercostal, lateral thoracic, and lumbar arteries, and hematoma usually developed along the chest wall or muscles (2,8).

However, to our knowledge, there have been no reports of spontaneous rupture of the internal thoracic artery or mediastinal hematoma in patients with cirrhosis. During the literature review, we found a single case report of a large anterior mediastinal hematoma due to spontaneous internal thoracic artery rupture in a patient with underlying lupus vasculitis (9); similar to our case, the patient was successfully treated with coil embolization.

In summary, we described spontaneous internal thoracic artery rupture in a patient with alcoholic liver cirrhosis. Asymptomatic arterial hemorrhage can develop because of liver cirrhosis without a traumatic event, which may require adequate intervention or surgical exposure.