Ann Thorac Surg 2003;75:593-596
© 2003 The Society of Thoracic Surgeons
Case report
Multiple pulmonary artery aneurysms due to infective endocarditis
A. Kür
at Bozkurt, MD*a,
Funda Öztunç, MDb,
Canan Akman, MDc,
Sebuh Kurugoglu, MDc,
Aye Güler Ero
lu, MDb
a Department of Cardiovascular Surgery, Istanbul University Cerrahpa
a Medical Faculty, Istanbul, Turkey
b Department of Pediatrics, Istanbul University Cerrahpaa Medical Faculty, Istanbul, Turkey
c Department of Radiology, Istanbul University Cerrahpaa Medical Faculty, Istanbul, Turkey
Accepted for publication August 14, 2002.
* Address reprint requests to Dr Bozkurt, Ataköy 5. K
sm A7/40 34750 Istanbul, Turkey
e-mail: akbozkurt{at}yahoo.com
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Abstract
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Bilateral pulmonary artery aneurysms developed in the course of staphylococcal endocarditis in a 6-year-old girl with ventricular septal defect. Consecutive computed tomography scans revealed the progressive enlargement of one of the aneurysms. She underwent an urgent left upper lobectomy because of the impending rupture and a possible life-threatening hemorrhage. The second ipsilateral aneurysm was plicated in order to exclude the aneurysm sac. In the next operation the ventricular septal defect was closed and vegetations located on the tricuspid valve were removed. On follow-up spontaneous thrombotic resolution occurred in the right-sided aneurysms.
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Introduction
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Since Osler’s original description in 1885 the term "mycotic aneurysm" has been used to indicate any infectious process involving the arterial wall [1]. Analysis of the data shows that such lesions have been observed very rarely in pulmonary arteries. They tend to grow rapidly with the risk of rupture and death [2, 3]. Surgical treatment is recommended whenever possible although spontaneous resolution has also been reported occasionally [4].
A 6-year-old girl was admitted after 10 days of high-grade fever, malaise, chills, and tachycardia. The family reported that she had not been "well" for almost 6 weeks although they did not seek medical attention during that period. On physical examination a systolic murmur was detected at the left sternal border. Chest roentgenogram showed a well-defined mass located at the left upper lobe and widening of both hilar areas. A helical computed tomography (CT) scan demonstrated a giant, lobulated-contoured left pulmonary artery aneurysm measuring 4 x 3.5 x 3 cm in size. Anterior to the aneurysm a parenchymal consolidation was noted. Another aneurysm was found at the left common basal pulmonary artery with the dimensions of 2 x 2 cm. Additionally there were two aneurysms located in the right middle lobe and right lower lobe pulmonary arteries that were measured at 1.5 x 1.5 cm and 2 x 2 cm in size, respectively (Fig 1).
Echocardiography demonstrated a perimembraneous ventricular septal defect (VSD) as well as vegetations on the septal leaflet of the tricuspid valve. Staphylococcus aureus was isolated from multiple blood cultures. We initiated vancomycin and the patient improved clinically after 2 weeks of treatment for subacute bacterial endocarditis. However, a repeat CT scan performed 18 days later revealed progressive enlargement of the aneurysm located in the left upper lobe to 5 x 4.5 x 5 cm, and also showed progressive subpleural extension (Fig 2).
The case was considered as "impending rupture" and urgent surgery was planned because of the probable life-threatening hemorrhage.
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Fig 1. Contrast-enhanced consecutive helical computed tomography scans reveal a giant left upper lobe pulmonary artery aneurysm (a). Surrounding lung parenchyma shows consolidation (bottom left panel). Note the additional left common basal (dash; bottom right panel) and right descending (circle; bottom left panel) small-sized aneurysms.
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Fig 2. Repeat computed tomography examination shows the enlargement and progressive subpleural extension of the left upper lobe pulmonary artery aneurysm (a: left and right upper panels).
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The patient underwent left posterolateral thoracotomy and a giant aneurysm originating from the truncal branch of the left pulmonary aneurysm was detected. The wall of the aneurysm had become thin. Segmental resection was not possible and left upper lobectomy was carried out. The second aneurysm was located at the anterior aspect of the left common basal artery. A straight vascular clamp was used in order to isolate half of the circumference of the pulmonary aneurysm. The aneurysm sac was buttressed with two, 2-cm–long bovine pericardium strips on either side of the pulmonary artery with 4-0 polypropylene sutures. Pathologic examination of the aneurysm wall demonstrated disruption of the intima and media. There was a lymphoplasmocytic inflammatory cell infiltration throughout the adventitia. Surrounding lung parenchyma showed organizing lipoid pneumonia. We did not observe any organisms in the specimen and the cultured sample of the aneurysm wall also revealed no organisms. Three weeks later the VSD was closed under cardiopulmonary bypass and the vegetations were removed. We explored the right lung but were not able to find the right-sided aneurysms because of the limited access through median sternotomy. The patient’s postoperative course was straightforward. She was kept on vancomycin for 6 weeks after the VSD closure. A CT scan performed 3 weeks after the second operation revealed almost complete thrombosis and regression of the right-sided aneurysms without additional aneurysm formation (Fig 3).
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Fig 3. Postoperative control computed tomography scans depict almost complete thrombosis of the right-sided aneurysms (white squares) and postoperative changes in the left hemithorax (left and right bottom panels).
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Comment
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Mycotic pulmonary artery aneurysm is a rare complication of right-sided endocarditis. In two collected series Charlton and associates [2] reported 16 cases between 1848 and 1961 and Benvenieste and coworkers [3] reported 25 cases between 1961 and 1988 in world literature. Most of the cases were associated with endocarditis due to congenital heart diseases or intravenous drug addiction. The clinical course of mycotic pulmonary artery aneurysms is not well documented. In the collective series reported by Benvenieste and colleaguges [3] mortality was rather high, as 14 of 23 patients in whom the outcome was specified died for a mortality rate of 54%. They recommended early embolization or surgery or both after diagnosis [3].
Behcet’s disease is a common vasculitis of unknown etiology in our country. Recurrent oral aphtous ulcers, genital ulcers, uveitis, and skin lesions characterize the disease. Large-vessel involvement may be observed in 15% to 35% of patients [5]. Pulmonary artery aneurysms may develop in the course of Behcet’s disease. Therefore Behcet’s disease was considered in the differential diagnosis of the reported case. However, according to the diagnostic criteria proposed by the International Study Group for Behcet’s disease, recurrent oral ulceration must be present as well as at least two of the following: recurrent genital ulceration, eye lesions, skin lesions, or a positive pathergy test. Our patient had none of these findings and Behcet’s disease was easily excluded [6].
The present case has several unique characteristics compared with previously reported patients in the literature. Pulmonary artery aneurysms developed multifocally and one of them enlarged within a short time, eventually necessitating an urgent operation. The 54% mortality rate reported by Benvenieste and coworkers [3] prompted us to consider urgent surgery. It is known that overflow in the pulmonary artery increases the risk of mycotic aneurysm [3]. Probably closure of the VSD accelerated the thrombosis and resolution of the right-sided pulmonary artery aneurysms and institution of an aggressive antibiotic therapy as well as cleaning the septic source with open-heart surgery prevented further aneurysm development.
The likelihood of spontaneous resolution might be expected for small mycotic pulmonary artery aneurysms but regular control CT scans are compulsory [4]. The timing of hemoptysis appears unpredictable and should alert the clinician to consider a more aggressive approach like surgery or embolization. Our recent experience with embolization of pulmonary artery aneurysms due to Behcet’s disease is favorable and may be considered as the first-line treatment modality [7]. However, in the present case subpleural extension of the thin-walled aneurysm would have caused intractable bleeding during an endovascular procedure. Surgical treatment should always be undertaken to prevent catastrophic consequences for large aneurysms. Direct ligation of the feeding vessel and endoaneurysmorrhaphy with preservation of lung tissue seems to be the ideal surgical procedure; however, it is rarely feasible as reported by Westaby and colleagues [8]. We believe that in appropriate cases the aneurysm sac could be safely excluded with bovine pericardium strips in order to preclude lung resection, as performed in the case described above.
This report draws attention to the occurrence and different clinical behavior of multiple pulmonary artery aneurysms in the same patient with bacterial endocarditis. Close follow-up with radiologic modalities demonstrated progressive enlargement of the left-sided aneurysms and surgical treatment prevented a possible fatal outcome. Conversely a conservative approach precluded an unnecessary operation on the right-sided aneurysms.
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References
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- Osler W. The Gulstonian lectures on malignant endocarditis. BMJ 1885;1:467-470.[Free Full Text]
- Charlton R.W., Du Plessis L.A. Multiple pulmonary artery aneurysms. Thorax 1961;16:364-371.
- Benvenieste O., Bruneel F., Bedos J.P., et al. Ruptured mycotic pulmonary artery aneurysm: an unusual complication of right-sided endocarditis. Scand J Infect Dis 1998;30:626-629.[Medline]
- McLean L., Sharma S., Maycher B. Mycotic pulmonary arterial aneurysms in an intravenous drug user. Can Resp J 1998;5:307-311.[Medline]
- Schirmer M., Calamia K.T., O’Duffy J.D. Is there a place for large vessel disease in the diagnostic criteria of Behcet’s disease?. J Rheumatol 1999;26:2511-2512.[Medline]
- International Study Group for Behcet’s Disease. Criteria for diagnosis of Behcet’s disease. Lancet 1990;335:1078-1080.[Medline]
- Bozkurt A.K. Embolisation in Behcet’s disease. Thorax 2002;57:469-470.[Free Full Text]
- Westaby S., Tan C., Foale R., Hallidie-Smith K.A., Bentall H.H. Mycotic aneurysm of the pulmonary artery. Direct surgical approach with preservation of lung tissue. J Cardiovasc Surg (Torino) 1987;28:596-598.[Medline]
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Abstract
Introduction
