Ann Thorac Surg 2008;86:655-657. doi:10.1016/j.athoracsur.2008.02.006
© 2008 The Society of Thoracic Surgeons
Case Reports
Successful Surgical Management of Invasive Aspergillosis of the Pulmonary Arteries
Giorgio Zanotti, MDa,*,
Salvatore Nicolardi, MDa,
Marco Morsolini, MDa,
Marco Maurelli, MDb,
Eloisa Arbustini, MDc,
Roberto Dore, MDd,
Andrea M. D'Armini, MDa
a Division of Cardiac Surgery, University of Pavia School of Medicine, Pavia, Italy
b Division of Critical Care Medicine and Anesthesia, University of Pavia School of Medicine, Pavia, Italy
c Department of Pathology, University of Pavia School of Medicine, Pavia, Italy
d Department of Radiology, University of Pavia School of Medicine, Pavia, Italy.
Accepted for publication February 4, 2008.
* Address correspondence to Dr Zanotti, Division of Cardiac Surgery, University of Pavia, Fondazione IRCCS Policlinico San Matteo, Piazzale Golgi 19, Pavia, 27100, Italy (Email: giorgiozanotti{at}hotmail.com).
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Abstract
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We describe an 11-year-old girl with severe obstruction of the main pulmonary arteries caused by invasive aspergillosis and managed with combined pulmonary endarterectomy and antimycotic treatment.
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Introduction
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Invasive pulmonary aspergillosis is a common complication among immunocompromised patients, and it is associated with high morbidity and mortality [1]. When invasive pulmonary aspergillosis is limited to the lung parenchyma, several surgical and medical options are available [2, 3]. However, when invasive aspergillosis involves the large vessels, outcomes are almost invariably dismal. Angio-invasive Aspergillus infection is resistant to antifungal therapy, becoming rapidly obstructive and lethal [4–6]. Arterial wall infection and frequent extension of the disease into more peripheral branches often preclude surgical resection and prosthetic graft interposition.
We report the successful treatment of histologically proven angio-invasive Aspergillus infection of the pulmonary arteries in a young patient with severe aplastic anemia. We chose a combined medical and surgical approach that included pulmonary endarterectomy (PEA).
An 11-year-old girl with a history of severe aplastic anemia presented to a community hospital with a fever and a nonproductive cough. A chest x-ray disclosed a left lower lobe consolidation, which was confirmed by computed tomography. Empiric broad-spectrum antibiotics and antifungal treatment (imipenem, teicoplanin, and amphotericin) was started, with rapid remission of her symptoms. Two weeks later, the patient became febrile and complained of dyspnea on mild exertion. A new-onset systolic heart murmur radiating to the back was detected on physical examination. Systolic pulmonary arterial pressure estimated by echocardiogram was 70 mm Hg. A chest computed tomographic angiogram showed occlusive and subocclusive filling defects of the left and right main pulmonary arteries without parenchymal pulmonary abnormalities (Fig 1). Bronchoalveolar lavage was unremarkable. The patient was also found to have an excessive factor VIII, von Willebrand factor, and plasminogen activator inhibitor. An endovascular biopsy of the vascular mass showed hyphae embedded in abundant thrombotic material. As platelet and neutrophil counts were 10 and < 1.5 x 103/mL, respectively, a trial of high-dose warfarin combined with amphotericin and voriconazole was undertaken. Despite initial improvement, the patient experienced chest pain and severe shortness of breath at rest within a couple of weeks. Systolic pulmonary pressure had risen to 90 mm Hg, and right ventricular systolic function appeared severely depressed on echocardiogram.
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Fig 1. The intravascular mass shows no contrast enhancement. The arterial wall of the left main pulmonary artery is vascularized and thickened. Lung parenchymal signs of invasive pulmonary aspergillosis or chronic thromboembolic pulmonary hypertension were absent.
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To restore patency of the pulmonary arteries and enucleate the infection, a bilateral PEA was emergently performed. A median sternotomy was used with aorto-bi-caval cannulation for cardiopulmonary bypass. Core body temperature was dropped to 28°C and the myocardium was protected by topical cooling and continuous cold blood (28°C) perfusion in the fibrillating heart. One single arteriotomy was performed from the distal part of the pulmonary trunk through the midportion of the left main pulmonary artery. A dissection plane was raised within the media of each of the main pulmonary arteries and was developed out to the takeoff of the lobar vessels. The right and left main pulmonary arteries and the artery to the left lower lobe were freed from all intravascular clots during three short periods of circulatory arrest (ie, 5 + 5 + 2 minutes). Because the lesion was invasive, a safe plane of dissection could not be raised on the artery to the left upper lobe, which was left occluded. The pathology report and cultures confirmed the presence of angio-invasive Aspergillus fumigatus. The postoperative course was uneventful. Platelets and granulocytes were transfused, and granulocyte colony-stimulating factor was administered. The patient was discharged on postoperative day 7 on an intravenous regimen of caspofungin, voriconazole, meropenem, teicoplanin and acyclovir. Eight weeks later, anti-infectious medications were discontinued. The girl fared well until 18 months later when she underwent chemotherapy and allogeneic bone marrow transplantation for leukemic transformation. The patient had no recurrence of fungal infection. She died of acute adenoviral hepatitis 22 months after surgery.
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Comment
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Invasive pulmonary aspergillosis is the second most common cause of death after leukemic transformation in patients with marrow aplasia [1], and invasion of the pulmonary arteries consistently leads to death [4, 5]. We believe that this is the first case of angio-invasive aspergillosis of the main pulmonary arteries successfully treated by pulmonary endarterectomy combined with systemic antifungal therapy. In our opinion, the key points of surgical treatment were endovascular enucleation with removal of the infected tissue out to the peripheral vessels, the lack of need for insertion of a vascular graft, and the perioperative systemic administration of multiple antifungal drugs. Main points to be pondered are the indications and the rationale for this novel surgical approach. In addition, the potential for cure must be weighed against the potential for fatal complications.
Hilar localization of Aspergillus infection and invasion of the great vessels almost invariably lead to death due to occlusion of the arterial lumen [4–6] or hemorrhage [2]. Emergent surgery was indicated because of the fast growing and obstructive nature of the intravascular lesions despite optimized medical treatment. In addition, because both main pulmonary arteries were proximally infected and no parenchymal lung involvement was detectable, none of the conventional surgical approaches [2, 3] was deemed suitable.
Pulmonary endarterectomy allows extensive removal of chronic thromboembolic material from the major pulmonary arteries. The chronic thromboembolic scaffold of the infection and its proximal localization made PEA technically ideal. Furthermore, PEA avoids unnecessary resection of lung parenchyma. Avoiding the creation of empty cavities not only decreases the risk of permanent pleural contamination in case of fungal dissemination, but also preserves lung function, which is of utmost importance in case of leukemic transformation and subsequent need for chemotherapy. Because no vascular resection is performed, the use of prosthetic material is avoided. Nevertheless, the ability of this novel surgical approach to achieve thorough enucleation of fungal infection had yet to be established. A few cases of favorable outcome with partial resection have been described [7].
The most dreadful complication of PEA is arterial wall perforation with small airway bleeding. Although the risk of perforation was certainly higher than in a normal PEA, it did not seem to outweigh the risk of imminent death from cardiac failure or sepsis in our case.
In this challenging case, the two main goals of therapy were relief from pulmonary hypertension and enucleation of the infectious process. With removal of the obstructing mycetoma, the patient experienced immediate relief from right ventricular overload with prompt and full recovery of right ventricular function. In addition, no recurrence of aspergillosis occurred in the patient during chemotherapy or after bone marrow transplantation.
Endarterectomy surgery may be a lifesaving procedure in selected cases of angio-invasive aspergillosis of the major pulmonary arteries complicating invasive pulmonary aspergillosis. Intravascular growth of the infection into surgically accessible vessels and the absence of parenchymal cavitary abscesses adjacent to the infected arteries may represent appropriate surgical indications for PEA in the setting of invasive pulmonary aspergillosis.
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References
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Abstract
Introduction
