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Allergic Bronchopulmonary Aspergillosis Mimicking Lung Cancer

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Allergic Bronchopulmonary Aspergillosis Mimicking Lung Cancer CASE REPORT Urszula Nowicka1, Elżbieta Wiatr1, Lilia Jakubowska2, Małgorzata Polubiec-Kownacka3 1Third Department of Lung Diseases, Institute of Tuberculosis and Lung Diseases, Warsaw, Poland Head of Department: Prof. K. Roszkowski-Śliż, MD, PhD 2Department of Radiology, Institute of Tuberculosis and Lung Diseases, Warsaw, Poland Acting Head of Department: I. Bestry, MD, PhD 3Endoscopy Laboratory, Department of Surgery, Institute of Tuberculosis and Lung Diseases, Warsaw, Poland Head: D. Dziedzic, MD, PhD Allergic bronchopulmonary aspergillosis mimicking lung cancer in a non-asthmatic female patient: a case report Alergiczna aspergiloza oskrzelowo-płucna imitująca guz płuca u chorej bez astmy oskrzelowej — opis przypadku Abstract Allergic bronchopulmonary aspergillosis (ABPA) is a lung disease caused by a hypersensitivity reaction to antigens of the Aspergillus species (most frequently Aspergillus fumigatus), with a variable radiographic appearance. ABPA most commonly affects patients with steroid-dependent asthma (1–2%) and patients with cystic fibrosis (5–15%). ABPA is very rarely diagnosed in non-asthmatics. We report a case of ABPA in a 45-year-old female initially evaluated for suspected cancer of the left lung with hilar lymphadenopathy, who had never been diagnosed with asthma. After the diagnostic investigation was complete, the diagnosis of ABPA was established and appropriate treatment was instituted leading to clinical, radiologi- cal, and serological improvement (IgE decrease). Key words: allergic bronchopulmonary aspergillosis (ABPA), asthma, lung cancer Introduction cough with expectoration of brownish purulent plugs and-less frequently-general symptoms, such Allergic bronchopulmonary aspergillosis as fever, weight loss, and asthaenia. ABPA is cha- (ABPA) is a disorder caused by a hypersensitivity racterised by a considerably variable radiographic reaction to antigens of the Aspergillus species (most appearance with the most common findings being frequently Aspergillus fumigatus). ABPA was first atelectasis, transient pulmonary infiltrates, proxi- described in 1952 by Hinson et al. [1]. The patho- mal bronchiectasis, and signs of mucoid impaction. genesis of ABPA is complex with immune and ge- We report a case of a previously healthy fe- netic factors on the part of the host being implica- male referred to our Department with suspected ted. ABPA most commonly affects patients with tumour of the left lung. allergic diseases, including steroid-dependent asth- ma (1–2% of ABPA patients) or cystic fibrosis (5– Case presentation 15% of ABPA patients) [2]. As ABPA is very rarely diagnosed in patients without a history of asthma, A 45-year-old female never-smoker with a no data on the incidence of the disease in the ge- long-standing history of allergy, receiving no long- neral population are available [3]. Patients usual- term medication, an ENT specialist, was admitted ly complain of paroxysmal dyspnoea, productive to our Department in December 2009 for diagno- Adres do korespondencji: Urszula Nowicka, MD, PhD, Third Department of Lung Diseases, Institute of Tuberculosis and Lung Diseases, Płocka St. 26, 01–138 Warsaw, Poland. tel.: +48 22 431 23 80; fax: +48 22 431 24 08; e-mail: [email protected] Manuscript received on: 30 June 2011 Copyright © 2012 Via Medica ISSN 0867-7077 www.pneumonologia.viamedica.pl 77 Pneumonologia i Alergologia Polska 2012, vol. 80, no 1, pages 77–81 stic evaluation of a left lung tumour. Since July 2009 she had been complaining of a cough with expectoration of mucopurulent sputum without haemoptysis, dyspnoea, or fever. Despite several courses of antibiotics (clarithromycin, cefuroxime, metronidazole) the cough kept returning after pe- riods of temporary improvement. In November 2009 a chest X-ray revealed partial atelectasis of the upper lobe of the left lung with sparing of the lingula, suggestive of a lung tumour (Figure 1). The chest CT scan performed at that time confirmed atelectasis of the upper lobe of the left lung. The patient underwent diagnostic evaluation for can- cer at an outpatient oncology facility, where she underwent flexible bronchoscopy revealing infil- tration of the bronchus and the carina leading to segment 1–2 of the left lung, pus filling the bron- chus, and infiltration of the bronchus leading to the lingula. Histopathological examination of the Figure 1. Postero-anterior X-ray of the chest — left upper lobe tissue samples collected from the left upper lobe atelectasis sparing lingula bronchus revealed massive inflammatory changes with necrotic masses with signs of low-degree dys- leading to the lingula. Microbiological examination plasia focally in the metaplastic epithelium. Ne- of the bronchial discharge revealed isolated fun- ither cytological examination of the bronchial wa- gal hyphae, and the cultures revealed numerous shings nor bronchial brushing revealed tumour Aspergillus fumigatus colonies and isolated Can- cells. Microbiological examination of the bronchial dida albicans colonies. No acid-fast bacilli were washings revealed a confluent growth of Aspergil- detected. The immediate aspergillin skin test was lus fumigatus and the presence of numerous Char- positive (5 × 5 mm) and the delayed test was ne- cot-Leyden crystals. The patient was then admit- gative. A positive result was obtained in the sero- ted to the Institute of Tuberculosis and Lung Dise- logical test for aspergillosis (IgG) and no IgE anti- ases in Warsaw for continuation of the diagnostic bodies specific for Aspergillus fumigatus were de- evaluation. tected. Spirometry did not reveal signs of airway On admission the patient was in a good gene- obstruction, although upon the administration of ral condition without cough, any other respirato- a short-acting b2-agonist a marked improvement of ry symptoms, or fever. She said she had lost about forced expiratory volume in one second (FEV1) was 5 kg in the past month. Chest auscultation reve- observed, which was considered a sign of bronchial aled crepitations over the anterior chest wall in the hyperreactivity. All these diagnostic investigations midclavicular line and anterior axillary line on the allowed us to rule out lung cancer and confirm the left. Differential blood cell count revealed an eosi- diagnosis of ABPA (the patient met 6 out of 8 ma- nophilia of 1.6 × 199/l (16.4%) and a marked ele- jor diagnostic criteria proposed by Rosenberg and vation of immunoglobulin E (IgE) to 1400 IU/ml. Patterson) [4, 5]. The patient was started on gluco- A chest X-ray revealed a widened outline of the left corticosteroids at the dose of 0.5 mg/kg/day, which hilum with atelectasis of the lingula (Figure 2). A were tapered off after 6 months, and on itracona- chest CT scan revealed proximal bronchiectasis in zole at a dose of 200 mg BID for 2 months (due to segment 3 of the upper lobe of the left lung and the massive fungal growth). the lingula with retention of discharge, causing A radiological improvement and a reduction partial atelectasis of the lingula and fine intralo- in serum IgE was observed during the follow-up bular nodules consistent with retained discharge after the initiation of treatment. in the small airways with reactively enlarged lymph nodes in the mediastinum (Figure 3). Com- Discussion pared to the previous radiograms, based on the variable presentation of the pulmonary changes, a Aspergillus is a common mould which acco- suspicion of ABPA was raised. Flexible broncho- unts for 0.1–22% of all fungal spores in air sam- scopy revealed severe inflammatory changes with ples. Although about 250 species of this fungus purulent discharge and plugging of the bronchus have been described so far, only some of them are 78 www.pneumonologia.viamedica.pl Urszula Nowicka et al., Allergic bronchopulmonary aspergillosis mimicking lung cancer in a non-asthmatic female patient in patients suffering from asthma or cystic fibro- sis, usually in the presence of atopy. The increased viscosity of the mucus in the respiratory tract in some of the patients with asthma and cystic fibro- sis combined with impaired mucociliary clearan- ce in cystic fibrosis disrupts the process of effecti- ve removal of the fungal spores from the bronchi. The susceptibility to the development of ABPA is also a result of genetic factors that determine in- flammatory response in atopic patients, which determines activation of T and B cells specific for Aspergillus [7, 8]. Five stages of the disease, which may develop in various orders, are distinguished: stage I (acute), stage II (remission), stage III (exa- cerbation), stage IV (steroid dependent), and stage V (fibrotic). The disease may be insidious with periods of exacerbation and periods of remission, and early diagnosis may prevent the progression of the disease, damage to the pulmonary intersti- Figure 2. Postero-anterior X-ray of the chest — partial regression tium, and pulmonary function deterioration [7]. of the upper lobe lesions, atelectasis of lingula appeared The major diagnostic criteria of ABPA inclu- de [4, 7, 8]: (1) asthma, (2) pulmonary infiltrates on radiograms, (3) positive immediate aspergillin skin test (type I hypersensitivity), (4) peripheral blood eosinophilia (> 1000/mm3), (5) serum pre- cipitins (IgG) to Aspergillus fumigatus, (6) total se- rum IgE exceeding 427 IU/ml (1000 ng/ml) [9, 10], (7) central bronchiectasis, and (8) serum IgE and/ or IgG specific for Aspergillus. The minor diagno- stic criteria of ABPA include the presence of Asper- gillus in the sputum, expectoration of brownish
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