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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 1  |  Page : 19-22

Subacute invasive pulmonary aspergillosis


Department of Pulmonary Medicine, DM Wayanad Institute of Medical Sciences, Wayanad, Kerala, India

Date of Submission12-Oct-2020
Date of Decision04-Nov-2020
Date of Acceptance08-Nov-2020
Date of Web Publication28-Jan-2021

Correspondence Address:
Dr. Ravindran Chetambath
Navaneeth, Sarovaram Road, Calicut - 673 020, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jalh.jalh_4_20

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  Abstract 


Subacute invasive pulmonary aspergillosis (IPA) is an indolent cavitating infectious process which can mimic other chronic lung infections such as tuberculosis. This usually occurs in mildly immunosuppressed individuals and clinically evolves over weeks to months. Radiological presentation is in the form of thick-walled cavity with or without mycetoma, fibrosis, and pleural invasion. Here, a case of subacute IPA in a middle-aged female with rheumatoid arthritis is reported, highlighting a rare radiological appearance.

Keywords: Aspergillosis, halo sign, pleural invasion


How to cite this article:
Chetambath R. Subacute invasive pulmonary aspergillosis. J Adv Lung Health 2021;1:19-22

How to cite this URL:
Chetambath R. Subacute invasive pulmonary aspergillosis. J Adv Lung Health [serial online] 2021 [cited 2021 Mar 4];1:19-22. Available from: http://www.jalh.com/text.asp?2021/1/1/19/308270




  Introduction Top


Subacute invasive pulmonary aspergillosis (IPA) previously called chronic necrotizing pulmonary aspergillosis (CNPA) or semi-IPA is an indolent cavitating infectious process which can mimic other chronic lung infections.[1] Diagnosis is often delayed and progression to chronic fibrosing pulmonary aspergillosis with irreversible lung damage is common. Subacute IPA usually occurs in mildly immunosuppressed individuals such as diabetes mellitus, chronic obstructive pulmonary disease, advanced age, malnutrition, alcoholism, connective tissue diseases, corticosteroid therapy, and radiation therapy.[2] Patients present with constitutional symptoms such as fever, malaise, fatigue, weight loss, cough, and hemoptysis. Radiologically, subacute IPA is characterized by pulmonary consolidations, usually in the upper lobes, progressing to cavitation over weeks to months associated with bronchiectasis.[3] Fungal ball with air crescent sign can occur due to parenchymal destruction by fungus.[4] Pleural extension with pleural fibrosis is also reported. A rare radiological finding is discussed here in a patient with rheumatoid arthritis presenting with chronic respiratory symptoms.


  Case Report Top


A 57-year-old female presented with symptoms of cough with scanty sputum for 1 year. She has exertional breathlessness and tiredness. There is no history of hemoptysis or chest pain. For these symptoms, she has consulted many times with only partial symptomatic relief. She is a known asthmatic since early adult life and was on inhaled long acting beta agonist and corticosteroid. She was not regularly using these drugs. She was diagnosed to have rheumatoid arthritis, 7 years back and was on methotrexate and pulse steroids. There was no history of antituberculous therapy. She was a hypertensive and not a diabetic. On examination, she was of average built having small joint deformity in both hands. There was bilateral wheeze.

Investigation showed a normal white blood cell count with elevated erythrocyte sedimentation rate (75 mm/1 h). Sputum for acid fast bacilli was negative on 2 occasions. Sputum KOH smear showed fungal mycelia. Fungal culture showed branching septate hyphae suggestive of Aspergillus species. Her collagen disease work up showed evidence of rheumatoid arthritis. X-ray chest posteroanterior view showed a cavity in the left upper zone with evidence of parenchymal fibrosis [Figure 1]. Computed tomography (CT) thorax showed multiple small well-defined thick walled cavities in the? Left upper lobe (LUL) with multiple nodular projections within [Figure 2]. One of the cavities showed air crescent sign [Figure 3]. There is extension to the neighboring pleura with pleural thickening and fibrosis. Cavity has an irregular appearance due to fibrosis. A diagnosis of subacute IPA was made based on the clinical presentation, presence of rheumatoid arthritis, use of immunosuppressants, radiological findings and isolation of Aspergillus fumigatus from the respiratory sample. Differential diagnosis of cavitating rheumatoid nodule was not considered due to the fungal growth in culture. The patient was treated with oral itraconazole and was advised to continue the drug for at least 6–8 weeks with regular follow-up to assess clinical response.
Figure 1: (a) X-ray Chest showing thick.walled cavity LUZ with evidence of fibrosis (Yellow arrows). (b) Computed tomography thorax showing pleural-based lesions with multiple small cavities, pleural thickening, and fibrosis (Yellow arrow head)

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Figure 2: (a) Multiple small cavities with fibrosis and pleural extension. One of the cavities show crescent sign. Pleural involvement and thickening resembles the base of a wine glass (Yellow arrows). (b) Showing a well-defined thick-walled cavity in the LUL with multiple nodular projections from the wall. Pleural extension also can be seen. Cavity wall is irregular due to fibrosis (yellow arrow head)

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Figure 3: (a and b) The “wine glass sign” in subacute invasive pulmonary aspergillosis formed by the parenchymal lesion and pleural extension. (b) Artistic design of a wine glass for comparison

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  Discussion Top


Fungal pneumonias though considered rather uncommon earlier are being diagnosed with increasing frequency. This is attributed to the increasing number of susceptible population as a result of widespread use of drugs such as antibiotics, corticosteroids, immunosuppressants, use of devices, organ transplantation, and acquired immunodeficiency. Increasing number of fungal infections in the pulmonary and critical care units has significantly affected the health-care cost in many countries. Mortality due to fungal infections is considered almost equal to that of tuberculosis and human immunodeficiency virus disease. Many people at risk for and suffering from fungal diseases live in resource-limited settings, where diagnosis and treatment of these infections can be challenging.[5]

Aspergillus is a ubiquitous saprophytic fungus commonly isolated from both the outdoor and indoor environment including hospitals. Heat, moisture, and organic matters favor its growth. A. fumigatus is the most common pathogen. Aspergillus terreus, Aspergillus niger, and Aspergillus flavus are the other important pathogens causing disease in man.[6] Aspergillus causes a variety of infections or allergic diseases in human hosts and the type of clinical disease depends on the host immune status and pulmonary structure.[1]

Aspergillus-associated lung diseases are grouped into different types.[3] In atopic individuals, the fungus triggers robust immune reactions and cause allergic rhinitis, asthma, hypersensitivity pneumonitis, and allergic bronchopulmonary aspergillosis. Saprophytic growth of Aspergillus in preexisting cavities gives rise to aspergilloma (fungal ball). Subacute IPA or CNPA occurs in patients with mild immunosuppression or chronic lung diseases.[2] IPA develops in severely immunocompromised patients.[7] These forms represent a continuum of disease and often, there is transition from one type to the other or overlap between these forms. The prevalence of Aspergillus species isolation in a cohort of hospitalized patient was 16.3% and the independent risk factor associated with Aspergillus infection in this cohort was diabetes mellitus which was clinically and statistically significant.[8]

Lesion typically involves the upper lobes and begins as a pulmonary opacity. Eventually, the central necrotic area separates away from the surrounding lung and thus forms an air crescent sign. This occurs in weeks to months, eventually resulting in a cavity with or without a central mycetoma.[4] Appearances may then be the same as an aspergilloma. Often there are multiple cavities which are thick-walled. Adjacent pleural thickening is often present. It is similar in appearance to tuberculosis, actinomycosis, and histoplasmosis.

Definitive diagnosis of IPA or CNPA depends on the demonstration of the organism in tissue by visualization of the characteristic fungi using Gomori methenamine silver stain or calcofluor; or positive culture result from sputum, needle biopsy specimen, or bronchoalveolar lavage (BAL). In biopsy specimens, hyphal invasion of lung parenchyma confirms diagnosis of subacute IPA. Bronchoscopic or open lung biopsy specimen may be subjected to fungal culture. BAL fluid for fungal stain and culture is generally helpful in the diagnosis. Sensitivity and specificity of BAL fluid is 50% and 97%, respectively. There are few previous studies, reporting different prevalence rates of fungal isolation in respiratory samples from patients with chronic obstructive pulmonary disease.[9],[10],[11] Recently, a large, retrospective study conducted by Guinea et al., analyzed the incidence of A. fumigatus isolation from lower respiratory tract samples in patients admitted for AECOPD to a tertiary hospital.[12] They reported 239 isolations of Aspergillus species (16.3 per 1000 admissions). Serum galactomannan testing is a useful test for early diagnosis and to monitor the disease during treatment. Sensitivity and specificity of galactomannan antigen assay in BAL fluid specimen is 77.2% and 77%, respectively, and in serum is 66.7% and 63.5%, respectively. The detection of (1-3)-β-D-glucan could be a promising nonculture based, noninvasive tool for the diagnosis of invasive aspergillosis.

The condition usually runs a slowly progressive course over weeks to months, and vascular invasion or dissemination to other organs is unusual. During initial phases of the disease, antifungals are the mainstay of treatment. Intravenous and intracavitary amphoterin B, 5-flucytosine, itraconazole and voriconazole have all been tried. Surgery is reserved for patients who do not respond to initial medical management and have an adequate pulmonary reserve and acceptable operative risks. Once the disease has ceased progressing treatment is the same as that of an aspergilloma. Prognosis is largely dictated by underlying lung disease and co-morbidities with mortality ranging from 10% to 39%.[13]

A diagnosis of subacute IPA was made in this case based on underlying mild immunosuppression due to rheumatoid arthritis, chronic respiratory symptoms, upper lobe cavity with halo sign, pleural invasion, and Aspergillus isolation from respiratory sample. Lung biopsy, which is the definitive test to demonstrate tissue invasion by fungal mycelia could not be performed in this case. Serum galactomannan also could not be done in this.

The highlight of this case is the CT Thorax findings depicting the cavities, mycetoma and pleural involvement acquiring the shape of a wine glass. The pleural extension and pleural fibrosis forms the base of the glass and the cavity and contents form the body of the glass. This is different from the wine glass appearance of allergic bronchopulmonary aspergillosis in which it is the V or Y shaped shadow due to mucus plugging of airways. This shadow is formed by parenchymal and pleural involvement in subacute IPA and is not described so far. This may be named as “wine glass sign,” a distinguishing feature in the diagnosis of subacute IPA.


  Conclusion Top


A case of subacute IPA in a patient with rheumatoid arthritis is presented here. Patient is on methotrexate and oral corticosteroid and is an ideal candidate to develop this disease. Detailed discussion of this condition is presented along with an interesting radiological sign which is named as “wine glass sign.” However, lung biopsy and serum galactomannan estimation if done could have given better evidence for the diagnosis.

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.



 
  References Top

1.
Kousha M, Tadi R, Soubani AO. Pulmonary aspergillosis: A clinical review. Eur Respir Rev 2011;20:156-74.  Back to cited text no. 1
    
2.
Saraceno JL, Phelps DT, Ferro TJ, Futerfas R, Schwartz DB. Chronic necrotizing pulmonary aspergillosis: Approach to management. Chest 1997;112:541-8.  Back to cited text no. 2
    
3.
Franquet T, Müller NL, Giménez A, Guembe P, de La Torre J, Bagué S, et al. Spectrum of pulmonary aspergillosis: Histologic, clinical, and radiologic findings. Radiographics 2001;21:825-37.  Back to cited text no. 3
    
4.
Yella LK, Krishnan P, Gillego V. The air crescent sign: A clue to the etiology of chronic necrotizing pneumonia. Chest 2005;127:395-7.  Back to cited text no. 4
    
5.
Global fungal diseases. Centers for Disease Control and Prevention. National Center for Emerging and Zoonotic Infectious Diseases (NCEZID). Division of Foodborne, Waterborne, and Environmental Diseases (DFWED). Available fr?om: www.cdc.gov/fungal/global/index.html. [Last accessed on 2020 Jul 31].  Back to cited text no. 5
    
6.
Agarwal R, Denning DW, Chakrabarti A. Estimation of the burden of chronic and allergic pulmonary aspergillosis in India. PLoS One 2014;9:e114745.  Back to cited text no. 6
    
7.
Chabi ML, Goracci A, Roche N, Paugam A, Lupo A, Revel MP, et al. Pulmonary aspergillosis. Diagn Interv Imaging 2015;96:435-42.  Back to cited text no. 7
    
8.
Lashmipriya S, Chetambath R, Sunny A, Shivashankaran S, Aslam M. Aspergillus spp. infection as a cause of acute exacerbations of chronic obstructive pulmonary disease: a prospective observational study. Int J Res Med Sci 2019;7:1604-9.  Back to cited text no. 8
    
9.
Borman AM, Palmer MD, Delhaes L, Carrère J, Favennec L, Ranque S, et al. Lack of standardization in the procedures for mycological examination of sputum samples from CF patients: A possible cause for variations in the prevalence of filamentous fungi. Med Mycol 2010;48 Suppl 1:S88-97.  Back to cited text no. 9
    
10.
Knutsen AP, Bush RK, Demain JG, Denning DW, Dixit A, Fairs A, et al. Fungi and allergic lower respiratory tract diseases. J Allerg Clinic Immunol 2012;129:280-91.  Back to cited text no. 10
    
11.
Agbetile J, Fairs A, Desai D, Hargadon B, Bourne M, Mutalithas K, et al. Isolation of filamentous fungi from sputum in asthma is associated with reduced post-bronchodilator FEV1. Clin Exp Allergy 2012;42:782-91.  Back to cited text no. 11
    
12.
Guinea J, Torres-Narbona M, Gijón P, Muñoz P, Pozo F, Pel¨ez T, et al. Pulmonary aspergillosis in patients with chronic obstructive pulmonary disease: Incidence, risk factors, and outcome. Clin Microbiol Infect 2010;16:870-7.  Back to cited text no. 12
    
13.
Denning DW, Cadranel J, Beigelman-Aubry C, Ader F, Chakrabarti A, Blot S, et al. Chronic pulmonary aspergillosis: rationale and clinical guidelines for diagnosis and management. Eur Respir J 2016;47:45.  Back to cited text no. 13
    


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