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Ann Thorac Surg 1999;67:1789-1791
© 1999 The Society of Thoracic Surgeons
a Divisions of Cardiology and Cardiothoracic Surgery, David Grant Medical Center, Travis AFB, Stanford, California, USA
b Department of Pathology, Stanford University, Stanford, California, USA
Accepted for publication November 9, 1998.
Address reprint requests to Dr Wong, Division of Cardiology, SGOMC, David Grant Medical Center, 101 Bodin Circle, Travis AFB, CA 94535
e-mail: andrew.wong{at}60mdg.travis.af.mil
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Introduction |
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A previously healthy 36-year-old man was seen in the emergency department with chief complaints of substernal chest pressure and shortness of breath for 1 hour with associated nausea, diaphoresis, and radiation to the left upper extremity. He had experienced similar symptoms on the preceding day while exercising. Review of systems was relevant for symptoms of influenza 3 weeks prior to presentation. He had no cardiac risk factors and no important medical or surgical history. The patient denied tobacco, alcohol, or illicit drug abuse and was not on a regimen of prescribed medications.
Physical examination revealed an athletic-appearing man who was diaphoretic and anxious. He was afebrile with a blood pressure of 144/94 mm Hg, heart rate of 90 beats per minute, respirations of 24/min with mild labor, and oxygen saturation of 88% to 92% on room air. There was no jugular venous distention. Pulmonary examination was notable for bibasilar crackles. Cardiac examination revealed a regular rhythm without rub or murmur. A fourth heart sound was present. The abdomen was benign without hepatomegaly. The extremities demonstrated no edema and normal pulses.
Routine laboratory studies including urine toxicology screen were unremarkable. Cardiac enzyme levels, electrocardiogram, and chest radiograph were also normal. An arterial blood gas study revealed a substantial alveolar-arterial oxygen gradient, and a subsequent ventilation/perfusion scan demonstrated a low probability for a pulmonary embolus.
The patient subsequently sustained a cardiac arrest with electrocardiographic data suggesting an acute anterolateral myocardial infarction. Aggressive resuscitative efforts included recombinant tissue plasminogen activator, dopamine hydrochloride, norepinephrine, and intraaortic balloon counterpulsation. The patient underwent emergent cardiac catheterization, which revealed a dramatic subtotal occlusion of the proximal left main coronary artery, which was not relieved by angioplasty (Fig 1). Thoracotomy revealed a cystic mass below the level of the carina, between the aortic root and the proximal left main coronary artery. The cyst was incised, releasing approximately 20 mL of a nonpurulent whitish fluid under pressure. The patient underwent coronary artery bypass grafting to the left anterior descending and left circumflex coronary arteries. He was placed on the heart transplant list but died of the disease during interim medical management. Postmortem examination revealed a cyst 8 x 3 x 4 cm located at the base of the heart, enveloped by epicardial tissue, and intimately associated with the great vessels (Fig 2). Extensive infarction of the left ventricular myocardium was observed. The coronary arteries exhibited mild eccentric fibrocalcific arteriosclerosis. Histologic assessment demonstrated respiratory epithelium consistent with an ectopic bronchogenic cyst (see Fig 2).
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Acute presentations resulting from BC in adults are unusual. Creech and DeBakey [4] reported ischemic electrocardiographic changes as a result of BC. Fratellone and colleagues [5] discussed a patient seen with hypotension, cyanosis, and pulsus paradoxus. Surgical exploration with resection was curative and demonstated a large BC impinging on the aorta, pulmonary vessels, and left atrium. Because of the risk of complications, the authors recommended elective surgical resection for BC if discovered in adults. Mather and associates [1] reported the case of a woman hospitalized for mechanical ventilation and inotropic support because of pulmonary edema resulting from compression of the pulmonary veins by a BC; she underwent subsequent surgical excision. In their series of 18 patients treated for BC, Patel and coauthors [6] had 2 patients with serious complications. Three patients who were initially asymptomatic ultimately required surgical intervention for symptoms, and the incidence of surgical complications was higher in patients who were symptomatic at the time of operation. These authors also advise surgical resection of all suspected BCs.
The mechanism underlying the pathophysiology in our patient is uncertain. It was apparent at cardiac catheterization and postmortem examination that the BC was compressing the left main coronary vessel. Despite the history of viral symptoms, the clinical evaluation failed to demonstrate an infectious etiology precipitating cyst enlargement. As the BC was filled with a nonpurulent whitish fluid, acute hemorrhage was not evident. There were no air-fluid levels radiographically and no obvious pulmonary communication at postmortem examination; thus a Valsalva-type mechanism of enlargement is also unlikely. In such a confined space as the mediastinum, it is possible that the cyst shifted or prolapsed to a position favoring compression of the coronary vessels. Alternatively, the process of slow growth could have led to the acute hemodynamic event, given the superimposed demands associated with intense exertion.
Bronchogenic cysts in the adult population can be symptomatic, but acute catastrophic presentations are rare. Nonetheless, there is growing evidence that BCs exhibit reasonable morbidity and can be lethal. This case demonstrates the importance of maintaining latitude in the differential diagnosis of chest pain in the previously healthy adult. Given the potential morbidity and mortality, whether symptomatic or incidental, the finding of a BC warrants routine surgical resection [3, 5, 6].
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