© 2004 by European Society of Cardiology
Copyright © 2003, The European Society of Cardiology
Native aortic valve vegetative endocarditis with Cunninghamella
Hospital of the University of Pennsylvania, 3400 Spruce Street, 9 Gates Pavilion, Philadelphia, PA 19103, USA
Received 16 May 2003; received in revised form 1 September 2003; accepted after revision 4 September 2003.
* Corresponding author. Tel.: +1-215-662-2285; fax: +1-215-349-8190. suttonm{at}mail.med.upenn.edu
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Abstract |
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Cunninghamella bertholletiae is a rare Mucor species that is seldom the cause for endocarditis of prosthetic heart valves. It is even a more uncommon cause of endocarditis of native heart valves. We present a case of Cunninghamella endocarditis of a native aortic valve in an immuno-compromised patient, diagnosed from tissue culture obtained at the time of surgery. There have only been two other reported cases of native valve endocarditis with a Mucor species, and in those cases, the diagnoses were made post-mortem.
Keywords: endocarditis; fungus; native-valve; mucormycosis; Cunninghamella
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1 Case |
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A 32-year-old gardener with a 15-year history of primary red cell aplasia requiring multiple blood transfusions and chronic corticosteroid therapy was admitted with left chest pain. His ECG was notable for sinus rhythm and lateral ST segment elevation. A transthoracic echocardiogram showed hypokinesis of the antero-lateral LV wall, moderate aortic insufficiency due to aortic leaflet thickening and a small pericardial effusion. Emergent left heart catheterization confirmed antero-lateral hypokinesis but demonstrated normal coronary arteries. A transesophageal echocardiogram showed confluent vegetative endocarditic thickening of the aortic valve leaflets, and a coating of vegetative material involving the aortic sinuses, surrounding the coronary ostia and extending for 3–3.5 cm up the ascending aorta (Fig. 1A–C). He was treated with amphotericin for culture-negative presumptive fungal endocarditis. He developed intractable congestive heart failure from rapidly progressive aortic regurgitation and underwent urgent aortic valve replacement and debridement of the proximal ascending aorta. Culture of the aortic valve revealed the Mucor species, Cunninghamella bertholletiae.
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Postoperatively he developed right foot drop and a left Horner's syndrome, and head CT scan revealed multiple embolic lesions. He subsequently developed complete heart block requiring pacemaker placement. He continued to have fevers on maximal doses of amphotericin and posaconazole. Repeat transesophageal echocardiogram two weeks postoperatively revealed recurrent confluent vegetations extending circumferentially throughout the aortic root and ascending aorta and a new mitral leaflet perforation with mitral regurgitation (Fig. 2A and B). He developed progressive heart failure, multiple organ failure and finally sustained a cardio-respiratory arrest.
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2 Discussion |
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Mucormycosis is a rare infection that typically affects immuno-compromised subjects. Cunninghamella bertholletiae is an atypical form of mucormycosis that occasionally causes isolated pulmonary disease.1 Cunninghamella is found in the soil in many temperate climates including the United States.1 The majority of patients affected by this organism are predisposed secondary to chemotherapeutic agents, chronic steroid use, hematological conditions requiring multiple blood transfusions and broad-spectrum antibiotics.1 Cardiac mucormycosis has been reported, but the majority of these cases were diagnosed at autopsy,2 and almost exclusively involved prosthetic valves.3 Only five patients with native valve endocarditis secondary to Mucor species have been described, all at autopsy, two involved the aortic valve and in none was the Mucor speciated.4–6
We report the first case of Cunninghamella aortic valve endocarditis diagnosed in life and confirmed by tissue culture. Our patient was immuno-compromised due to chronic corticosteroid therapy, received multiple blood transfusions for red cell aplasia and had daily exposure to soil working as a gardener. This case is noteworthy for the unusual echocardiographic appearance of the vegetations, which spread up the endocardial surface of the aorta from the aortic valve surrounding the ostia of the coronary arteries, and their early and aggressive reappearance postoperatively resulting in multiple systemic emboli. The presenting chest pain syndrome, ECG changes, concordant regional wall motion abnormalities were likely due to coronary embolism. The very unusual confluence of minimally mobile vegetations demonstrated echocardiographically in culture-negative endocarditis unresponsive to antifungal agents should raise the possibility of Mucor endocarditis.
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References |
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- Ribes A.R., Vanover-Sams C.L., Baker D.J. Zygomycetes in human disease. Clin Microbiol Rev (2001) 13:236–301.[CrossRef][Web of Science]
- Virmani R., Connor D.H., McAllister H.A. Cardiac mucormycosis: a report of five patients and a review of 14 previously reported cases. Am J Clin Pathol (1982) 78:42–47.[Web of Science][Medline]
- Ellis M.E., Al-Albedy H., Sandridge A., Greer W., Ventura W. Fungal endocarditis: evidence in the world literature, 1965–1995. Clin Infect Dis (2001) 32:50–62.[CrossRef][Web of Science][Medline]
- Gloor F., Loffler A., Scholer H.J. Mucormycosen. Pathol Microbiol (1961) 24:1043–1064.
- Straatsma B.R., Zimmerman L.E., Gass J.D.M. Phycomycosis: a clinicopathologic study of fifty-one cases. Lab Invest (1962) 11:963–985.[Web of Science][Medline]
- Jackman J.D., Simonsen R.L. The clinical manifestations of cardiac mucormycosis. Chest (1992) 101:1733–1736.[CrossRef][Web of Science][Medline]
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