A Case series of ABPA with rare radiological presentation

Sudhir Kumar; Deependra Kuamr Rai; Subhash Kumar

doi:10.4103/ijaai.ijaai_19_20

CASE REPORT

Year : 2020 | Volume : 34 | Issue : 2 | Page : 117-122

A Case series of ABPA with rare radiological presentation

Sudhir Kumar, Deependra Kuamr Rai, Subhash Kumar
Department of Radiology, AIIMS, Patna, Bihar, India

Date of Submission	02-May-2020
Date of Acceptance	25-Jun-2020
Date of Web Publication	20-Nov-2020

Correspondence Address:
Dr. Deependra Kuamr Rai
Department of Radiology, AIIMS, Patna, Bihar
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/ijaai.ijaai_19_20

Abstract

Allergic bronchopulmonary aspergillosis (ABPA) is an immunologically mediated lung disease that occurs in response to Aspergillus fumigatus which affects primarily asthmatic and cystic fibrosis patients. Central bronchiectasis is a classical radiological presentation of ABPA, but the presentation as total collapse of the lung, pneumothorax, lung mass, or fibrocavitary disease has been rarely described. First case was a 50-year-old nonsmoker male who presented radiologically as complete collapse of left lung. The second case was 54-year-old nonsmoker male presented with breathlessness and chest radiograph showed right-sided pneumothorax which expand after chest tube insertion. The third case a 35-year-old female who treated as bronchial asthma and refer to us for provisional diagnosis of malignancy after saw chest radiograph. The last case was a 22-year-old male who had typical symptoms and radiologically features of pulmonary tuberculosis. All these patients were treated by antitubercular drug, but later, all four case turn out as ABPA. This case series highlights the rare radiological presentation of ABPA (pneumothorax, lung collapse, lung mass, and fibrocavitary), and identifying these diseases in the early stage can prevent the development of end-stage pulmonary fibrosis.

Keywords: Allergic bronchopulmonary aspergillosis, lung collapse, pneumothorax, pulmonary tuberculosis

How to cite this article:
Kumar S, Rai DK, Kumar S. A Case series of ABPA with rare radiological presentation. Indian J Allergy Asthma Immunol 2020;34:117-22

How to cite this URL:
Kumar S, Rai DK, Kumar S. A Case series of ABPA with rare radiological presentation. Indian J Allergy Asthma Immunol [serial online] 2020 [cited 2021 Nov 28];34:117-22. Available from: https://www.ijaai.in/text.asp?2020/34/2/117/300918

Introduction

Allergic bronchopulmonary aspergillosis (ABPA) is a hypersensitivity reaction in response to colonization of the airways with Aspergillus fumigatus that occurs commonly in patients with asthma or cystic fibrosis (CF).^[1],[2] In chronic cases, repeated episodes of bronchial obstruction, inflammation, and mucoid impaction can lead to bronchiectasis, fibrosis, and other respiratory complication.^[3] The clinical picture of ABPA is dominated by asthma and recurrent exacerbations. In severe cases, episodes of bronchial obstruction, fever, malaise, expectoration of brownish mucus plugs, and sometimes hemoptysis can occur. Based on serology and radiological findings, the disease is classified as ABPA-S (serologic) or ABPA-central bronchiectasis (CB), respectively, depending on the absence or presence of bronchiectasis or ABPA-CB-other radiologic findings (ORF).^[4] A recent study by Kumar and Goel found a relative incidence of ABPA, ABPA-CB, and ABPA-ORF to be 22.32%, 43.75%, and 33.92%, respectively.^[5]

The chest radiographic findings depend on the stage of the disease. During acute exacerbations, transient and fleeting opacities are characteristically found, while fixed abnormalities are seen in chronic stages of the disease. The common findings include consolidation, mucoid impaction of bronchi, and areas of atelectasis, whereas rare findings include pleural effusion, perihilar bronchocele mimicking adenopathy or mass lesion, miliary nodules, pneumothorax, and cavity and unilateral lung collapse. Chest radiographic findings are generally nonspecific, and high-resolution computed tomography (HRCT) of the chest is the imaging of choice in ABPA. The findings on the chest HRCT include CB, mucus plugging with bronchocele formation, and others.^[6] ABPA is rarely present with unilateral complete lung collapse, pneumothorax, cavitary consolidation, and as dilated bronchus appears as space-occupying lesion (SOL). We report here four cases of ABPA with rare radiological finding mentioned above.

Case Reports

Case 1: Allergic bronchopulmonary aspergillosis with complete lung collapse

A 50-year-old nonsmoker male presented with dry cough and breathlessness for 1 year and worsened for the last 2 days. On examination, vitals were within normal limit, except saturation dropped to 92% at room air. Chest auscultation showed absent air entry on the left hemithorax. The patient was using oral corticosteroid and bronchodilator (tablet betnesol and theo-asthalin) as on need basis, and he was also put on antitubercular treatment for the last 2 months by a general physician. There was no history of hemoptysis, chest pain, wheeze, fever, anorexia, or weight loss. Chest X-ray showed complete collapse of left lung with compensatory emphysema on the right hemithorax [Figure 1]. Computed tomography (CT) chest showed along with collapse of the left lung, cylindrical bronchiectasis in the right perihilar region, and hyperattenuating mucus plug in the left main bronchus [Figure 2]a and [Figure 2]b. Video-bronchoscopy performed on the same day showed thick tenacious secretion in the left main bronchus [Figure 3]. Bronchial secretion was aspirated for fungal hyphae; acid–fast bacilli (AFB) stain come out as negative. On blood investigations, the patient had a total leukocyte count (TLC) of 11,300 cells/mm³ with 76% neutrophils, 18% lymphocytes, and 05% eosinophils. Total serum immunoglobulin E (IgE) level estimated using fully automated chemiluminiscence system was 19,803 IU/mL (<100 IU/mL). Specific IgE against A. fumigatus was 19.40 kU/L (<0.1 kU/L). Patient was diagnosed as ABPA with left lung collapse and he was put on oral prednisolone 0.75 mg/kg with inhaled corticosteroid and bronchodilator. After 2 weeks, chest radiograph showed complete resolution of collapse lung [Figure 4].

Figure 1: Frontal view chest radiograph showing white left hemithorax with transmediastinal herniation of right lung and upward pull of left hemidiaphragm and stomach

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Figure 2: Computed tomography scan of chest (a) lung window and (b) soft tissue window showing left lung collapse, bronchiectasis in right lung, and dense radiopaque material filling the left main and distal bronchi

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Figure 3: Video bronchoscopy showing thick tenacious secretion at left main bronchus completely occluding the left main bronchus

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Figure 4: Follow-up frontal view chest radiograph showing re-expanded left lung with few patchy opacities in the left mid-zone and mild hyper expansion of the left upper zone

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Case 2: Allergic bronchopulmonary aspergillosis with pneumothorax

A 54-year-old nonsmoker, diabetic male presented with exertional dyspnea, chest pain, fever, and anorexia for the last 2 months. There was a history of recurrent, cough, and dyspnea for the last 15 years. The patient was also put on antitubercular treatment for the last 9 months by a general physician. On examination, vitals are pulse 100/min, blood pressure (BP) 130/80 mmHg, respiratory rate (RR) 24/min, afebrile, and oxygen saturation 96% at room air. Chest radiograph showed pneumothorax right lung with passive collapse of the lung [Figure 5]a which expanded after chest tube placement [Figure 5]b. After few months, chest X-ray done showed bilateral radiopaque opacity in the mid-zone [Figure 5]c, which revealed bilateral cystic bronchiectasis and hyperattenuated mucus plug (HAM) in the HRCT chest scan [Figure 6]a and [Figure 6]b. Blood investigation revealed 12% eosinophilia with serum-specific IgE against A. fumigatus 5.15 KU/L (<0.1 KU/L) and serum total IgE 5545.5 IU/mL (<100 IU/mL). Spirometry showed forced vital capacity (FVC) of 3.48 L (106% of predicted), a forced expiratory volume in 1 s (FEV1) of 2.27 L (89% of predicted), and an FEV1/FVC ratio of 65.1%; there was insignificant bronchodilatation after administering a bronchodilator. The patient was put on oral corticosteroid with inhaled corticosteroid, and bronchodilator leads to better symptom control and resolution of shadow present in the chest X-ray [Figure 5]d.

Figure 5: Serial chest radiographs (a) right pneumothorax (b) linear opacities in the right mid-zone (c) patchy opacities in the right mid-zone and perihilar region (d) irregular and linear shadows in right middle zone and nodular opacities in both lungs in the lower zones

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Figure 6: High resolution computed tomography scan of chest (a) axial section at level of aortic arch, showing multiple mucus filled dilated bronchi on the right side, multiple centriacinal nodules with tree in bud configuration on both sides (b) axial section at lower level, showing right middle lobe collapse-consolidation with bilateral bronchiectasis and multiple centriacinar nodules

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Case 3: Allergic bronchopulmonary aspergillosis with lung mass

A 35-year-old nonsmoker female presented with increased in breathlessness, cough, and fever for the last 2 months. She had exertional dyspnea and recurrent symptoms which get worsen in the last 5 years. The patient was treated as bronchial asthma and receiving inhaled bronchodilator and corticosteroid on basis. On examination, her vitals were within normal limit. Chest examination showed bilateral wheeze and coarse crepitation. Chest radiograph shows well-defined radiodense opacity in the right upper zone, mimicking as mass lesion with patchy consolidation in the left lower zone [Figure 7]. CT of the chest showing a lobulated mass in the right upper lobe, ground-glass opacities, and hyperattenuating mucus plug seen in the anterior part of the lesion [Figure 8]a and [Figure 8]b. Spirometry performed showed FVC of 2.23 L (78% of predicted), a FEV1 of 1.45 L (64% of predicted), and an FEV1/FVC ratio of 65.02%; there was significant bronchodilatation (18% and 270 ml) after administering a bronchodilator. Blood investigation revealed 19% eosinophilia with serum-specific IgE against A. fumigatus 17.4 kU/L (<0.1 KU/L) and serum total IgE 11245 IU/mL (<100 IU/mL).

Figure 7: Frontal chest radiograph showing well defined water density mass in the right upper zone and patchy opacities in the left lower zone

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Figure 8: Computed tomography of chest (a) lung window and (b) soft tissue window showing a lobulated mass in posterior segment of right upper lobe, mild surrounding ground glass opacities are seen and hyperattenuating mucus plug is seen in the anterior part of the lesion

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The patient was given oral corticosteroid and inhaled corticosteroid and bronchodilator, but posttreatment chest radiograph was not available as the patient could not turn up during the follow-up.

Case 4: Allergic bronchopulmonary aspergillosis with multicavitary consolidation mimicking as tuberculosis

A 22-year-old nonsmoker male presented with fever, cough, and blood in the sputum for the last 20 days. He gave a history of recurrent cough and SOB during coughing for the last 5 years but never received inhaler. There was history of antitubercular treatment 1 year back for 6 months. On examination, pulse was 88 per min, BP 100/50 mmHg, RR 28/min, and SpO₂ 97% at room air. Chest auscultation showed bilateral wheeze and crepitation in the right hemithorax. Chest X-ray showed patchy infiltrate and consolidation in the right upper zone [Figure 9]. CT scan showed consolidation with ground opacity surrounding (halo sign) and bilateral cystic bronchiectasis [Figure 10]. Sputum microscopy for AFB and GeneXpert was negative. Blood investigation showed Hb 11.40, TLC 11900, neutrophil 36%, lymphocyte 40%, and eosinophil 20%. Serum-specific IgE against A. fumigatus 2.1 kU/L (<0.1 KU/L) and serum total IgE 1527 IU/mL (<100 IU/mL). Spirometry indices were suggestive of asthma. The patients was put on standard ABPA treatment which showed complete resolution of chest X-ray opacity [Figure 11].

Figure 9: Frontal chest radiograph showing right upper lobe collapseconsolidationwith multiple cystic lesion, widened right paratracheal stripe, and left mid and lower zone infiltrates

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Figure 10: High-resolution computed tomographic scan, axial section, showing consolidation in the posterior segment of the right upper lobe with surrounding ground glass opacities; bilateral bronchiectasis is present with multiple centriacinar nodules with tree in bud appearance

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Figure 11: Follow-up frontal chest radiography after treatment for allergic bronchopulmonary aspergillosis, showing resolution of most of the shadows with some fibrotic lesions in the right upper zone

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Discussion

ABPA is an inflammatory pulmonary disorder known to complicate the course of bronchial asthma and CF. Natural course of ABPA is not well defined but often indolent and may be present for years before diagnosis. The disorder occurs secondary to the immune response against antigens released by A. fumigatus, which colonizes the airways of these patients. The diagnosis of ABPA is currently made on a combination of clinical, radiological, and immunological findings using the Patterson criteria.^[7] The diagnostic criteria for ABPA are divided into major and minor ones. The major criteria includes asthma, fleeting shadows on chest radiograms, immediate cutaneous reactivity to A. fumigatus, elevated total serum IgE, precipitating antibodies against A. fumigatus, peripheral blood eosinophilia, elevated serum IgE and IgG to A. fumigatus, and central/proximal bronchiectasis with normal tapering of distal bronchi. The minor criteria are expectoration of golden brownish sputum plugs, positive sputum culture for Aspergillus species, and delayed skin reactivity to A. fumigatus. If six of the eight major criteria are met, the diagnosis is said to be made with certainty. The most common chest radiographic finding noted is a normal chest radiograph. Consolidation is considered to be the most frequent chest radiographic abnormality of ABPA.^[6] Other chest radiographic features include transient pulmonary infiltrates (mainly in the upper and lower lobes), atelectasis, band-like opacities (the gloved finger sign), bronchiectasis, and less frequently areas of poor vascular pattern, cavities, and signs of fibrosis.^[8] We report here four cases of ABPA with rare radiological presentation. The characteristics of four cases are summarized in [Table 1]. Our first case presented with complete collapse of the lung with respiratory failure which later diagnosed as ABPA. Berkin et al.^[9] in 1982 described four patients with complete or partial atelectasis without a history of asthma in whom the final diagnosis was ABPA. A case of total collapse of the right lung in a patient with ABPA has been reported from Japan by Nomura et al. in a 29-year-old man with bronchial asthma.^[10] Agarwal et al. described a similar case of a 22-year-old female without any prior respiratory symptom history who presented with respiratory failure secondary to left lung collapse.^[11] In 2015, Kumar et al. also showed three cases of total or lobar collapse of lung.^[12]

Table 1: Characteristics of allergic bronchopulmonary aspergillosis (with atypical presentation)

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We report a second case of ABPA with the right side pneumothorax which improved after chest tube drainage, oxygen, and bronchodilator. Standard ABPA treatment was also started. There are few case reports of pneumothorax available in the literature. Our literature search yielded three case reports of pneumothorax in ABPA. Ricketti et al. were the first to describe a case of secondary spontaneous pneumothorax (SSP) in a patient with ABPA.^[13] They hypothesized that the SSP was a consequence of underlying bullous lung disease and that this finding is rare in ABPA because of the central distribution of the disease. Judson et al. have reported a case of SSP associated with BPF in a patient with ABPA that is the only previous case with the finding of BPF.^[14] They have suggested that the formation of the subpleural cystic spaces may be secondary to ball valve obstruction of the small airways by mucus. Another case report of right upper lobe collapse and right-sided pneumothorax on separate occasion in an 18-year-old male, suffering from asthma with ABPA.^[15]

Our third case of a 50-year-old nonsmoker male who presented radiological as lung mass mimicking as lung cancer who turn out to be dilated bronchus with HAM. Similar case described by Sanchez-Alcaros et al. in a 65-year-old male smoker without a history of asthma who had undergone thoracotomy due to suspected lung cancer, which revealed the presence of dilated bronchi filled with mucus and was diagnosed as ABPA.^[16] Kannan et al. showed a 55-year-old nonsmoker presented with nonhomogenous opacity in the left mid-zone suspected as lung cancer, later on diagnosed as ABPA.^[17]

In areas with a high prevalence of pulmonary tuberculosis (PTB), patients with ABPA having atypical radiological presentations are often misdiagnosed with PTB. In some Indian studies, ABPA was misdiagnosed as PTB in as high as 17%–50% cases and treated with antitubercular drugs for a long time.^[18] There are several case reports of ABPA mimicking as PTB available in the literature,^[19] but our case is different who presented with various radiological sigs such as multiple fibrocavitary lesion in the right upper zone, tree in bud, fleeting shadow. Ground glass opacity and halos sign was also present mimicking as pulmonary tuberculosis and invasive fungal infection respectively. In our case series, all the patients were labeled as PTB initially and received antitubercular treatment. The constitutional symptoms such as fever, cough, and loss of appetite which is classical of PTB were also found in ABPA.

Conclusion

This case series highlights that ABPA can have rare radiological presentations such as complete collapse of the lung, pneumothorax, SOL, and fibrocavitary lesion. Hence, keeping the varied radiological presentations in our mind, we need to be very vigilant to keep differential as ABPA in all these radiological situations to detect disease in the early stage and prevent the development of end-stage pulmonary fibrosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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