Infection DOI 10.1007/s15010-013-0575-z

CASE REPORT

Chronic necrotizing pulmonary aspergillosis in an immunocompetent patient: report of a rare case E. Jin • L. M. Wang • Q. Y. Li • X. Feng S. L. Ma



Received: 22 October 2013 / Accepted: 14 December 2013 Ó Springer-Verlag Berlin Heidelberg 2013

Abstract Chronic necrotizing pulmonary aspergillosis (CNPA) is a relatively uncommon manifestation of infection with Aspergillus spp. which mainly affects immunocompromised or immunostressed individuals with underlying lung diseases. Here, we present a case of mediastinum-involved CNPA in an immunocompetent patient with no symptoms and previous good health. Keywords Chronic necrotizing pulmonary aspergillosis  Immunocompetent  Case report  Computed tomography

Diagnosis relies upon radiographic findings of a progressive infiltrate with pleural thickening in the upper lung lobes [4]. Very few cases of disseminated aspergillosis in immunocompetent individuals have been reported in the literature [5]. Here, we describe a rare case of CNPA in an immunocompetent patient with no symptoms and in general good health. Computed tomography (CT) scan performed in a routine physical examination revealed a masslike consolidation without cavitation in the right lower lobe, and the diagnosis was confirmed by histological analysis and microbiological culturing.

Introduction Case report Chronic necrotizing pulmonary aspergillosis (CNPA) is one type of chronic pulmonary aspergillosis (CPA) and a relatively uncommon manifestation of infection with Aspergillus spp. [1] but which mainly affects immunocompromised [2] or immunostressed individuals with underlying lung diseases [3], such as tuberculosis.

E. Jin  L. M. Wang Department of Respiratory Medicine, Nanjing Medical University Affiliated Hangzhou Hospital (Hangzhou First People’s Hospital), Huansha Road 261, Hangzhou 310006, China Q. Y. Li Department of Pathology, Nanjing Medical University Affiliated Hangzhou Hospital (Hangzhou First People’s Hospital), Huansha Road 261, Hangzhou 310006, China X. Feng  S. L. Ma (&) Department of Medical Oncology, Nanjing Medical University Affiliated Hangzhou Hospital (Hangzhou First People’s Hospital), Huansha Road 261, Hangzhou 310006, China e-mail: [email protected]

A 43-year-old woman was referred to our hospital to address a tumor-like shadow discovered by chest roentgenogram during routine examination. Before hospital admission, the patient had no symptoms. The patient had no history of alcohol abuse or smoking. Physical examination revealed good general health status, with no overt signs of disease. The routine panel of laboratory tests provided unremarkable findings, with white blood cell count, C-reactive protein level and erythrocyte sedimentation rate within normal range. A tuberculin skin test (purified protein derivative) was also negative. Blood test for antibodies to the human immunodeficiency virus (HIV) was negative. CT scan of the chest showed a 3.1 9 4.5 cm mass adherent to the diaphragm, with pleural thickening in the right lower lobe (Fig. 1). After fiberoptic bronchoscopy revealed no abnormal findings, a CT-guided aspiration specimen was obtained from the lesional area in the right lower lobe. The specimen was greenish in color and contained no tissue. Microbiological evaluation of the aspirate showed Aspergillus

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Fig. 2 Histological analysis of the surgical biopsy using PAS staining. a Numerous Aspergillus hyphae in the lumen of bronchus (black arrows); 9400 magnification. b Numerous Aspergillus hyphae in the small airway mucosa invasion (black arrows); 9400 magnification

Fig. 1 CT scan of the chest demonstrating right lower lobe nodular opacities. a Lung window setting showing a 3.1 9 4.5 cm nodular opacity with regular margin adhesions to the diaphragm and pleural thickening in the right lower lobe. b Mediastinal window setting showing a low attenuation signal suggestive of a hypodense sign in the mass

fumigatus-positive culture and staining. Potential differential diagnoses were pulmonary aspergillosis and lung cancer complicated by aspergillosis. The patient insisted on the lobectomy, citing her reasoning as both her mother and husband having died of lung cancer. A rightlower lobectomy was performed and revealed a

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5 9 6 9 7 cm mass with dense adhesions to the histological examination of the resected specimen showed multiple small ill-defined granulomatous areas and areas of necrosis that were rimmed with histiocytes, giant cells, and lymphocytes, and had adjacent fibroplastic demarcations. These necrotic zones showed positive periodic acid-Schiff (PAS) staining for both septate hyphae (Aspergillus spp.) and for small airway mucosa invasion (Fig. 2). According to the histological and culture findings, the diagnosis of CNPA was made. After the right lower lobectomy, IgA, IgG, IgM, C3, C4, and blood TBNK lymphocyte subsets were analyzed, and all found to be within normal limits. The patient was immediately started on a 3-month course of oral itraconazole. Follow-up at 12 months showed no signs of recurrence and the patient reported continued general good health with no complaints.

Chronic necrotizing pulmonary aspergillosis

Discussion The spectrum of pulmonary aspergillosis manifestations is wide and relatively non-specific, ranging from cough, fever, malaise, fatigue, hemoptysis, shortness of breath, and unintentional weight loss. Currently, five clinical types of Aspergillus infection are recognized: tracheobronchial aspergillosis, allergic bronchopulmonary aspergillosis, mycetomas, CNPA, and invasive aspergillosis [6]. CNPA is among the rarest of the types, and is characterized as a slowly progressive form of pulmonary aspergillosis. Recently, a general consensus in the fields of CPA research and clinical management has emerged in which the disease is classified as CNPA or CCPA. The majority of CNPA cases arise as a complication of pre-existing chronic lung diseases, such as emphysema, sarcoidosis, tuberculosis sequelae, and other fibrotic lung diseases. An altered systemic immune status, such as that due to diabetes mellitus, chronic liver disease, malnutrition, connective tissue diseases, alcoholism, and low-dose corticosteroid therapy, is also considered a risk factor for CNPA [1]. While immunocompromise or immunostress predisposes an individual to Aspergillus spp. infection [7], immunocompetent individuals are also susceptible [8, 9]. However, diagnosis of CNPA under immunocompetent conditions is difficult, due to the immune system’s compensatory resolution of those symptoms seen in the immunocompromised patients. Regardless of the sufferer’s immune status, diagnosis of CNPA relies on clinical and radiographic findings. The most common radiological profile includes chronic fibrocavitary infiltrates, progressive cavitations, and pleural thickening in the upper lung lobes [10]. However, the suggestive finding in the current case of a healthy, immunocompetent woman (with no history of alcohol abuse or smoking) was a nodular opacity on X-ray. Such a case is rare, and the diagnosis could have been easily missed or delayed without the subsequent CT investigation that showed the mass-like consolidation without cavitations in the right lower lung lobe or the histological analysis that showed parenchymal invasion. Neither of these findings is typical of CNPA. Considering the fact that the current patient had no basic diseases, pulmonary or otherwise, and that the CNPA occurred in the lower lung lobe, it is possible that the disease may have been associated with longterm inhalation of spores—similar to the etiology of aspiration pneumonia. Other clinical findings that support this theory are the culturable A. fumigatus from the aspirate specimen and the histological evidence of fungal invasion into the lung tissue. Lung biopsy specimens are generally obtained through transbronchial biopsy or transthoracic puncture [11]. Surgical biopsy is rarely performed in CNPA patients, because of the increased risks associated with impaired respiratory function or severity of the

patient’s comorbidity. In the current case, it was impossible to definitely rule out the possibility of malignancy for the nodular opacity based on the CT findings alone [12, 13]. Lung biopsy was essential to differentiate between lung cancer and the unique form of pulmonary aspergillosis that was eventually confirmed by histology and culturing. Thus, this is the first case report in which CNPA parenchymal invasion was found by a surgical biopsy obtained during a right lower lobectomy. Itraconazole is a potent antifungal agent that has good safety profile and is effective against CNPA as both oral and parenteral preparations [14]. A completed itraconazole regimen is considered essential to avoid recurrence of CNPA [15]. The 3-month course of oral itraconazole administered to the current patient likely helped to facilitate the disease-free status that was maintained up to the 1-year follow-up.

Conclusion To the best of our knowledge, this is the first reported case in the literature of CNPA occurring in an immunocompetent host with previous good health, no comorbidities, and no overt signs of disease. Surgical treatment followed by a course of antifungal drug administration effectively resolved the condition and could be a good choice for treatment of CNPA in immunocompetent patients. Conflict of interest On behalf of all authors, the corresponding author states that there is no conflict of interest.

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Chronic necrotizing pulmonary aspergillosis in an immunocompetent patient: report of a rare case.

Chronic necrotizing pulmonary aspergillosis (CNPA) is a relatively uncommon manifestation of infection with Aspergillus spp. which mainly affects immu...
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