Embolization for haemomediastinum due to spontaneous rupture of oesophageal branch arising from an unusual origin (inferior thyroidal artery) after vomiting: a case report

Introduction

Spontaneous haemomediastinum is a rarely observed and potentially life-threatening condition.^1,2 A sudden increase in the intrathoracic pressure, such as that caused by coughing, sneezing or vomiting, or sudden sustained hypertension, can lead to mediastinal haemorrhage.^1–3 Among mediastinal haemorrhages, those in the pulmonary and bronchial arteries are the most frequently reported. However, reports of the oesophageal artery as the source of the haemomediastinum are extremely rare.^4,5 Therefore, awareness of the possibility of bleeding from rare sources is important. Transcatheter arterial embolization (TAE) is considered the best treatment option for mediastinal haemorrhage and has recently been introduced as a possible safe and efficacious treatment option for this rare condition.^1–4

This current case report describes a male patient with ruptured oesophageal branches arising from the inferior thyroidal artery (ITA) and bronchial artery after vomiting, which caused a massive mediastinal haemorrhage. The patient was successfully treated using TAE with N-butyl cyanoacrylate (NBCA). To the best of our knowledge, this is the first time a spontaneous rupture of an oesophageal branch arising from the ITA has been reported.

Case report

In 2019, a male in his mid-70s presented to the emergency room of Chungbuk National University Hospital, Cheongju-si, Korea with a 2-h history of severe sudden onset chest pain preceded by vomiting after a large meal. His past medical history was significant for chronic obstructive pulmonary disease; however, he had no history of trauma. Upon arrival, he was conscious; his blood pressure was 90/65 mmHg and his heart rate was 110 beats/min. His peripheral artery oxygen saturation was 90% on room air and 5 l of oxygen was administrated via nasal cannula to maintain sufficient oxygen saturation. Physical examination was normal. Laboratory workup revealed decreased haemoglobin level (11.5 g/dl). The vital signs stabilized after fluid resuscitation. Enhanced chest computed tomography (CT) revealed a focal wall defect of the upper thoracic oesophageal wall at the level of the first thoracic vertebra (T1) with middle mediastinal air-fluid collection, suggesting oesophageal perforation (Figure 1(a)), while the chest CT, obtained at a different level, also revealed a large haematoma surrounding the collapsed oesophagus in the middle mediastinum with internal extravasation of contrast medium, suggesting active bleeding (Figure 1(b)). The active bleeding was identified connected to a small artery that branched from the right bronchial artery. It was assumed that the small branch was an oesophageal artery based on its origination and distribution. Therefore, emergency angiography was performed. Selective right bronchial artery angiography using a 5 Fr cobra catheter (Cook Medical Inc, Bloomington, IN, USA) confirmed a pseudoaneurysm in an oesophageal artery that had an anastomotic branch with the bronchial artery (Figure 1(c)). A 1.7 Fr. microcatheter (Asahi Inc, Aichi, Japan) was advanced near the bleeding site and TAE was successfully performed using a 1:2 mixture of NBCA and iodized oil, which resulted in complete embolization. Eight hours after the first TAE, the patient’s blood pressure dropped and his haemoglobin level declined from 10.3 g/dl to 7.3 g/dl. A follow-up CT scan revealed residual active bleeding within the previous haemomediastinum (Figure 2(a)). The patient underwent repeat angiography. Given the follow-up CT finding in which the perforation site involved the upper portion of the thoracic oesophagus, it was predicted that the oesophageal branch from the ITA could be an additional bleeding focus. Therefore, a 5-Fr catheter was inserted into the right subclavian artery and a microcatheter was advanced into the right thyrocervical trunk using the coaxial technique. Selective angiography of the ITA showed extravasation of contrast media from the oesophageal artery arising from the descending branch of the ITA (Figure 2(b)). Embolization was performed with a 1:2 mixture of NBCA and iodized oil, which resulted in complete embolization and resolution of the extravasation. Contrast oesophagogram after TAE showed an intrathoracic extra-oesophageal leak of the contrast into the mediastinum from the right aspect of the upper third of the oesophagus (Figure 3(a)). Open surgery was not considered as a treatment option because the patient had a high risk of perioperative death and limited life expectancy. Subsequently, an emergent oesophagogastroduodenoscopy was performed 8 h after symptom onset. This examination revealed a huge tear at the right lateral aspect of the proximal oesophagus, approximately 20 cm from the incisor and a haematoma (Figure 3(b)) that was successfully controlled by a full-coverage oesophageal stent. The patient was discharged with no further treatment for the mediastinal bleeding.

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Figure 1.

Imaging findings for a male in his mid-70s who presented with a 2-h history of severe sudden onset chest pain preceded by vomiting after a large meal: (a) enhanced chest computed tomography (CT) showed a focal wall defect of the upper thoracic oesophageal wall (arrow) at the level of the first thoracic vertebra (T1) with the middle mediastinal air-fluid collection, suggesting oesophageal perforation (asterisk); (b) chest CT, obtained at a different level, revealed a large hematoma surrounding the collapsed oesophagus in the middle mediastinum with internal extravasation of contrast medium, suggesting active bleeding and (c) selective right bronchial artery angiography confirmed a pseudoaneurysm (arrow) in an oesophageal artery that had an anastomotic branch with the bronchial artery.

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Figure 2.

Imaging findings for a male in his mid-70s who presented with a 2-h history of severe sudden onset chest pain preceded by vomiting after a large meal: (a) a follow-up computed tomography scan revealed residual active bleeding within the previous haemomediastinum and (b) superselective angiography of the inferior thyroidal artery with microcatheter showed extravasation of contrast media (thick arrow) from the oesophageal artery arising from the descending branch of the inferior thyroidal artery (thin arrow).

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Figure 3.

Imaging findings for a male in his mid-70s who presented with a 2-h history of severe sudden onset chest pain preceded by vomiting after a large meal: (a) contrast oesophagogram showed an intrathoracic extra-oesophageal leak of the contrast (arrow) into the mediastinum from the right aspect of the upper third of the oesophagus and (b) emergent oesophagogastroduodenoscopy was performed, which revealed a huge tear (arrow) at the right lateral aspect of the proximal oesophagus from 20 cm to 27 cm from the incisor. The colour version of this figure is available at: http://imr.sagepub.com.

The institutional Review Board of Chungbuk National University Hospital approved the retrospective review of the patient’s medical records (no. 2023-01-014-005) and waived the requirement for obtaining informed consent form the patient. All patient details were de-identified and the patient provided written informed consent for all treatments. The reporting of this case report conforms to CARE guidelines. ⁶

Discussion

Oesophageal perforation is a rare condition that is commonly misdiagnosed and it can be fatal if not promptly treated. ⁷ Iatrogenic causes, such as paraesophageal surgery or oesophagoscopy, account for 59% of cases.^4,7 The second leading cause is spontaneous rupture (15%).^4,7 It usually results from a sudden increase the intraoesophageal pressure. These conditions occur during straining, coughing, weightlifting, and vomiting, as shown in this current case.^3,4,7 This perforation usually involves the left posterolateral aspect of the distal intrathoracic oesophagus; however, it can occur in the cervical or intra-abdominal oesophagus as well. ⁸ This current report presents a unique case of high intrathoracic oesophageal rupture after vomiting with an associated complication of haemomediastinum. Diagnosis of oesophageal perforation can be difficult because of the nonspecific symptoms. Contrast swallow examination and upper oesophageal endoscopy are diagnostic modalities of choice in case of suspicion of oesophageal rupture. CT scans have become very useful in the diagnosis of oesophageal perforations that are missed on oesophagography. Furthermore, their sensitivity and specificity in detecting mediastinal emergencies, such as bleeding secondary to oesophageal perforation, can reach up to 100%, even in early clinical presentations. ⁷

Various causes of acute haemomediastinum are often life-threatening. These include cases of trauma, tumours, iatrogenic procedures and ruptured aneurysms.^1–3 Case reports about haemomediastinum caused by ruptured aneurysms are not new; however, almost all previously reported cases describe pulmonary and bronchial artery aneurysms.^1–3 After reviewing the existing literature (in English), only one case of a ruptured aneurysm of the proper oesophageal artery confirmed by angiography was identified, which resulted in symptomatic mediastinal haematoma. ⁴ To the best of our knowledge, this current case report is the first to describe a haemomediastinum caused by oesophageal branch rupture arising from the ITA. The arterial blood supply of the oesophagus is divided into three parts depending on the oesophageal level. ⁹ Primarily, the descending branches of the ITA supply the cervical oesophagus. Branches of the subclavian, common carotid or superior thyroid arteries may also supply the cervical oesophagus. Branches of the bronchial arteries, and one or two oesophageal arteries that arise directly from the aorta, supply the midthoracic portion. Lastly, branches of the left gastric or left inferior phrenic arteries supply the distal portion. Therefore, a proper standardized angiography protocol for oesophageal arterial bleeding should be based on the anatomical understanding of the arterial supply to the oesophagus to save time and reduce radiation exposure as well as to minimize the recurrent bleeding that can be caused by collateral channels. This current case supports the notion that, for proximal oesophageal perforations associated with arterial bleeding, subclavian and carotid arteriograms should also be evaluated.

With the advancement of microcatheter and embolic agents, TAE has quickly become a safe and effective treatment for many cases of oesophageal bleeding.^9,10 The various types of embolic materials used for the embolization of the oesophageal artery include gelatin sponge particles, polyvinyl alcohol particles and micro-coils. As the oesophageal arteries form collateral capillary networks before they penetrate the oesophageal muscle layer and perfuse the submucosa, they can cause back bleeding (haemorrhaging from potential collateral channels), which is of concern. In this current case, NBCA was used as the embolic agent, which can penetrate potential collateral channels and effectively embolize these channels with a single injection due to its liquid characteristics. Recently, NBCA has been used as an embolic agent in patients with oesophageal bleeding and has shown promising results as observed in this current case. ¹⁰ However, there is a concern of the danger of oesophageal necrosis due to ischaemic injury and the difficulty in handling the NBCA material. To prevent this complication, an appropriate volume, speed of injection and adjustable ratio of iodized oil to NBCA mixtures should be used. In this current case, a 1:2 mixture of NBCA and iodized oil was used, which was carefully injected under fluoroscopic monitoring.

In conclusion, haemomediastinum is a rare complication with several possible causes including ruptured oesophageal artery associated with oesophageal perforation. Therapeutic angiography is emerging as an effective and safe treatment strategy for oesophageal arterial bleeding. Increased awareness of bleeding from unusual oesophageal arteries, such as branches from the ITA, would decrease the rates of technical failure or rebleeding in any radiological procedure.