Asian Cardiovasc Thorac Ann 2005;13:374-376
© 2005 Asia Publishing EXchange Ltd
Secondary Rupture of Aorta Following the Surgical Management of Aortoesophageal Fistula
Tao Jin, MD,
Guo-Wei Yu, MD,
Liang Ma, MD
Department of Cardiothoracic Surgery The First Affiliated Hospital Medical College of Zhejiang University Zhejiang, China
For reprint information contact: Tao Jin, MD Tel: 86 571 8723 6841 Fax: 86 571 8723 6628 Email:tjincn{at}21cn.com, 79 Qingchun Road, Hangzhou, Zhejiang 310003, China.
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ABSTRACT
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A patient suffering from an aortoesophageal fistula (AEF) caused by a fish bone, was treated in our institute in 2000. The operation was successful and the patient had an uneventful early postoperative course. However, the patient died of frank hematemesis on the 6th postoperative day due to secondary rupture of the aorta. The lessons learnt and surgical efforts to manage AEF caused by an esophageal foreign body are discussed.
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INTRODUCTION
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Aortoesophageal fistula is a rare but life-threatening disorder. The etiologies of AEF in decreasing order of frequency are (1) aortic aneurysm, (2) neoplasm (especially carcinoma of the esophagus), and (3) miscellaneous abnormalities including esophageal foreign bodies, inflammatory diseases of the esophagus, and the dehiscence of a prosthetic aortic graft.1–2 Foreign bodies in the esophagus occur more often in children than in adults, yet rarely cause an AEF. It is postulated that either direct penetration or pressure necrosis of the esophageal wall initiates the fistula. Subsequent sepsis (mediastinitis and localized aortitis) eventually results in fistula formation.
The first case of AEF was reported by Dubrueil in 1818. The remarkably consistent clinical picture was first clearly described by Chiari in 1914 and confirmed in subsequent cases reported in the literature. To date, Chiari’s triad of midthoracic pain, sentinel hemorrhage, and a symptom-free interval followed by fatal exsanguination, is still well regarded as the most important clinical finding. There have been only a few survivors in about 100 reported AEF cases caused by an esophageal foreign body. The first recorded survival was reported by Ctercteko and Mok3 in 1980. It was not until the advent of cardiovascular and bypass procedures that such cases evolved from being pathological curiosities to treatable lesions.
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CASE REPORT
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A 49-year-old male was admitted on 30 October 2000. He had swallowed a fish bone a week prior to admission and now presented with pain in the mid-retrosternum, which was exacerbated by swallowing. He also complained of repeated hematemesis, of up to 600 mL of blood in the preceding week. The patient did not suffer from any pre-existing gastroesophageal disease. Roentgenography revealed no foreign body, but an emergency upper endoscopy disclosed an ulcer on the lateral wall of the esophagus, 26 cm from the incisor. Active bleeding was observed from the ulcer, but no fish bone was found. Based on this, a diagnosis of AEF was made. A massive hematemesis (more than 1 liter) occurred about 4 hours after the original sentinel hemorrhage. An emergency left thoracotomy was performed while on deep hypothermia cardiopulmonary bypass support, and a perforation, 5 mm in diameter, was found on the medial side of the aorta. This was repaired with a 4/0 Prolene running suture. A 4-mm-hole in the esophagus was oversewn with 4/0 Maxon running sutures. No pus and only a small amount of granulation and fibrinous tissue were observed. The fish bone was not found. The repaired esophagus and aorta were isolated by an autologous pericardial patch and a pedicle pleura flap. The microbiological study of the sample from the perforated area yielded a mixed growth of aerobes and anaerobes. The patient did well until the 6th postoperative day when he had a sudden circulatory collapse after frank hematemesis. He died of exsanguination 20 minutes later. A secondary rupture of the aorta was thought to be the most likely cause of death.
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DISCUSSION
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Table 1
illustrates the variety of methods that have been used to treat AEF and the patients’ outcome. As shown, despite the different surgical approaches, the overall survival rate is low. In 100 reported AEF cases caused by foreign bodies, only 12 have survived. To date, there has only been one reported case of secondary aortic rupture following the surgical management of an AEF.4 Surgical efforts were unsuccessful.
The surgical management of AEF involves the following challenges: first, control of the hemorrhage; second, arterial reconstruction in the infected area; third, control of sepsis; fourth, prevention of a secondary rupture of the aorta; and finally, re-establishment of the alimentary tract.
The symptoms of AEF are known as Chiari’s three signs. According to past reports, the time period from the initial bleeding to the massive bleeding ranges from 2 hours to 3 weeks (mean 5–7 days). In exceptional cases, it may last as long as 4 years. This period is shorter in cases of direct penetration of the aortic wall. During this interval the patient may have persistent symptoms of painful dysphagia and low-grade fever, or remain asymptomatic.
With the development of medical technology, the early diagnosis of AEF has become easier. If a patient vomiting blood visits a hospital, he or she usually undergoes an emergency endoscopy to identify the site and cause of bleeding. Although endoscopy is essential for suspected foreign bodies in the esophagus, the procedure itself has its own risks and can lead to hemorrhage. With the help of computed tomography (CT), we can construct a three-dimensional image to reveal the detailed relationship of the foreign body with the aorta. The initial management of a foreign body in the esophagus remains a problem. Once the AEF is diagnosed, the foreign body should not be removed. The blood vessel most usually perforated by a foreign body in the esophagus is the aorta, 1 to 5 cm from the origin of the left subclavian artery. This is the narrowest point of the esophagus, where foreign bodies are more likely to be impacted, and where the aorta and the esophagus are in closest proximity.
During the preoperative period, blood transfusion and fluid resuscitation are sufficient to stabilize the circulation. Any unnecessary vasoconstrictors should be avoided as their use can lead to uncontrollable hematemesis. A Sengstaken-Blakemore tube can also be inserted to tamponade the esophagus and control the hemorrhage. This has proven to be an effective method leading to good outcomes.
An operation should be performed without any delay once signs of hemorrhage are observed in AEF patients. A left thoracotomy is the preferred approach, as this gives the best access to the site of the fistula, as well as control of the aorta above and below the area. Cardiopulmonary bypass is essential for conducting a safe procedure.
In the postoperative period, a secondary rupture of the aorta is seldom reported. Wilson4 may have been the first such report. The patient died of secondary rupture on the 29th postoperative day, even though a flap of intercostal muscle was used to reinforce the aortic wall.
In our case, the problem may have resulted from a small esophagostoma, aortitis or mediastinitis. Therefore, intensive care is highly recommended in order to find a possible aortic aneurysm in the early period immediately after primary surgery. To monitor the developing changes in the mediastinum and aorta, bronchoscopic ultrasonography (BUS), chest CT or MRI is suggested in the days after the operation. We recommend a chest CT or BUS on the second day after the operation and then every 3 days in the first couple of weeks. If an aortic aneurysm is discovered and the infection is not severe, a secondary operation to resect the aortic aneurysm will be necessary to avoid a secondary rupture. Even if BUS, CT or MRI do not find an aortic aneurysm, a secondary operation to resect the concerned aorta is still highly recommended, to prevent a case of sudden death, as occurred in Wilson’s4 report.
In our case, although the patient’s temperature and white blood cell were normalizing postoperatively and there were no clear indications of infection before the patient’s collapse, we still believe that the secondary rupture of the aorta was caused by local infection which led to reopening of the aortic hole. As a result, we recommend performing a thoracic esophagectomy when there are concerns regarding contamination. This has been supported by the majority of the reported cases resulting in patient survival (Table 1
). The gastric tube with omentum may either be able to replace the esophagus or prevent mediastinal infection. From this case, we believe that the use of an esophagoscope after the primary operation, maybe on the 7th postoperative day, is necessary to find small esophagostoma.
Based upon the lessons learnt and successful experience in the management of AEF due to an esophageal foreign body, we conclude that: (1) a secondary rupture of the aorta is likely to have a fatal outcome in patients who have received surgical management of AEF due to an esophageal foreign body; (2) close follow-up, BUS, chest CT or MRI and the use of an esophagoscope in the early postoperative period are necessary in order to find possible secondary aortic changes in such patients; and (3) an esophagectomy with esophagogastromy for the contaminated esophagus is highly recommended to prevent esophageal infection.
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REFERENCES
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- Han SY, Jander HP, Ho KJ. Aortoesophageal fistula. South Med J 1981;74:1260–2.[Medline]
- Carter R, Mulder GA, Snyder EN Jr, Brewer LA 3rd. Aortoesophageal fistula. Am J Surg 1978;136:26–30.[Medline]
- Ctercteko G, Mok CK. Aorto-esophageal fistula induced by a foreign body: the first recorded survival. J Thorac Cardiovasc Surg 1980;80:233–5.[Abstract]
- Wilson RT, Dean PJ, Lewis M. Aortoesophageal fistula due to a foreign body. Gastrointest Endosc 1987;33:448–50.[Medline]
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ABSTRACT
INTRODUCTION
