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J Thorac Cardiovasc Surg 2008;136:1091-1093
© 2008 The American Association for Thoracic Surgery
Brief Communication |
a Department of Thoracic and Cardiovascular Surgery, The Cleveland Clinic Foundation, Cleveland, Ohio
b Metrohealth Medical Center, Cleveland, Ohio
Received for publication November 15, 2007; accepted for publication November 25, 2007. * Address for reprints: Karl G. Reyes, MD, Department of Thoracic and Cardiovascular Surgery, The Cleveland Clinic Foundation, 9500 Euclid Ave H35, Cleveland, OH 44195. (Email: reyesk2{at}ccf.org).
Aortoesophageal fistula (AEF) is rare and often fatal complication of vehicular trauma. In the past, it has also been reported in the presence of congenital arch anomalies. We report here a case of traumatic AEF in a patient with both a right-sided aortic arch and a Kommerell diverticulum, the first such in the literature, as well as its successful management by staged open surgical repair.
A 26-year-old man was taken to the emergency department after a vehicular crash in which he had been the ejected, unrestrained driver. Injuries noted on admission were primarily intracranial, with a depressed mental status necessitating mechanical ventilation. The patient's mental status eventually improved to the point at which he could be weaned from the ventilator and required only a percutaneous endoscopic gastrostomy tube for feeding. He was discharged to a rehabilitation center 3 weeks later.
During the patient's stay at the rehabilitation center, approximately 4 weeks after the crash, he had massive gastrointestinal bleeding, with bright red blood coming out of his percutaneous endoscopic gastrostomy tube. The patient was taken to a nearby hospital, where he was resuscitated and subsequently taken to the endoscopy suite. On esophagogastroscopy, large amounts of blood and blood clots were seen, but with no identifiable source of bleeding. Because of the suddenness of the event, the diagnosis of AEF was considered, and the patient was flown to our institution. On arrival, the patient had no active bleeding. Diagnostic studies included chest radiography, results of which were unremarkable except for findings suggestive of a right-sided aortic arch. Chest CT scan (
Figure 1) confirmed the presence of a right-sided aortic arch and descent of the aorta on the right side of the chest. In addition, there was an aberrant left subclavian artery with an aneurysmal origin, consistent with a Kommerell diverticulum, and an anteriorly displaced esophagus that narrowed at the level of the aberrant left subclavian artery. The suspected AEF, however, was not seen on the CT scan. Esophagogastroscopy was repeated and this time showed a nonbleeding, pulsating, 0.7-cm ulcer within the esophagus at 25 cm, corresponding to the level of the aortic knuckle. The patient was then taken to the operating room.
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The patient's postoperative course was unremarkable. He was extubated the day after his second operation. His mental status remained essentially unchanged from admission, and 11 days after the second operation, he was discharged to the rehabilitation facility.
The patient was seen again 10 days later for outpatient follow-up. He appeared to have recovered well from his surgery, and another CT scan showed no evidence of disruption of either repair. Subsequent follow-up calls during the next couple of months yielded positive reports, with the patient continuing to recover neurologically. His percutaneous endoscopic gastrostomy tube was eventually removed, and he was eventually able to tolerate a normal diet. There have been no further episodes of gastrointestinal bleeding. It is now 5 years since the crash. The patient is alive and well, fully functional, and seeking employment.
AEF was first described by Dubrueil1 in 1818 after a soldier swallowed a piece of rib, which perforated his descending thoracic aorta and eventually caused his death. Since then, there have been only a few reported cases in the literature of AEF secondary to trauma, and only 1 other in a patient with a right-sided aortic arch.2 Most AEFs reported in the literature have typically been related to ruptured thoracic aneurysms. Hollander and colleagues3 described 500 cases of AEF, and descending thoracic aneurysms accounted for 51% of the cases. Other causes included foreign-body perforations, thoracic malignancies, and postoperative complications; these made up approximately 40% of the cases. The remaining described cases of AEF were caused by esophageal reflux, tuberculosis, corrosive esophagitis, congenital anomalies, atherosclerotic disease, or instrumentation. There were only 6 reported cases (1%) of traumatic AEF in Hollander and colleagues' series.3
The diagnosis of AEF is often made at clinical presentation. Survival is rare, because most patients die immediately. If the patient survives the sentinel bleeding, diagnostic evaluation should follow immediately. Emergency endoscopy is often the initial diagnostic test. In stable patients, other diagnostic tests, such as CT scan and aortography, are reasonable for confirmation. In unstable patients, immediate surgical management is the only option. The tenets of successful surgical treatment are control of hemorrhage with repair of the aortic injury and individualized management of the esophagus, depending on patient stability and the underlying esophageal pathologic condition. The injury to the esophagus varies, from a simple, small perforation that requires primary repair to a completely destroyed esophagus with a contaminated thorax and mediastinum that requires esophagectomy and staged reconstruction. Any suture lines mandate interposition flaps. Broad-spectrum antibiotics are used to control infection resulting from esophageal leakage into the chest and mediastinum.
There has been only 1 other case reported in the literature of AEF in a patient with a congenital right-sided aortic arch, and no previous reports of AEF in a patient with both a right-sided aortic arch and Kommerell diverticulum. Kommerell diverticulum (
Figure 2) is a saccular aneurysmal dilation at the origin of an aberrant right or left subclavian artery. It is a rare anomaly that occurs in association with a double or left aortic arch and an aberrant right subclavian artery (0.5% of the population) or with a right aortic arch and an aberrant left subclavian artery (0.1%). Embryologically, it represents the persistent distal end of the interrupted fourth aortic arch, between the carotid and subclavian arteries.4 The question arises as to whether this congenital anomaly increases the likelihood of development of an AEF in response to disease or disruption. Kommerell diverticulum represents a likely area of weakening in the aortic wall that can also compress the esophagus and, in theory, could cause erosion of the structure into each other, especially when disruption of adventitial layers has occurred, such as with trauma.
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References
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