The Journal of Association of Chest Physicians

CASE REPORT
Year
: 2018  |  Volume : 6  |  Issue : 1  |  Page : 21--25

Spectrum of Misdiagnosis of Allergic Bronchopulmonary Mycosis: Case Reports


Sandeep Rana 
 Department of Respiratory Medicine, Military Hospital, Dehradun, Uttarakhand, India

Correspondence Address:
Sandeep Rana
Department of Respiratory Medicine, Military Hospital, Dehradun 248003, Uttarakhand
India

Abstract

Allergic bronchopulmonary mycosis (ABPM) is most commonly seen in cystic fibrosis and bronchial asthma. Most of patients are misdiagnosed at initial outpatient department visit due to low suspicion, lack of awareness of entity and similarity in clinical symptoms and radiological findings with other common pulmonary diseases. Pulmonary tuberculosis is the most common entity diagnosed, and anti-tubercular treatment is the most commonly abused chemotherapy. Careful history taking, astute initial examination and awareness may help in curbing wrong diagnosis and treatment. This article present two cases of ABPM who were initially misdiagnosed as pulmonary tuberculosis and carcinoma lung, respectively.



How to cite this article:
Rana S. Spectrum of Misdiagnosis of Allergic Bronchopulmonary Mycosis: Case Reports.J Assoc Chest Physicians 2018;6:21-25


How to cite this URL:
Rana S. Spectrum of Misdiagnosis of Allergic Bronchopulmonary Mycosis: Case Reports. J Assoc Chest Physicians [serial online] 2018 [cited 2023 Jan 28 ];6:21-25
Available from: https://www.jacpjournal.org/text.asp?2018/6/1/21/220988


Full Text



 Introduction



Allergic bronchopulmonary mycosis (ABPM) is idiopathic inflammatory allergic reaction to various fungi, which are ubiquitous in nature. This is cumulative type 1 and type 3 response to antigens.[1] Bronchial asthma and cystic fibrosis are the most common diseases associated with ABPM. Aspergillus fumigatus is the most common type of fungus involved but Candida albicans, Curvularia lunate, Saccharomyces cerevisiae, Penicillium sp., etc. are also involved. This was first described by Hinson et al. in 1952. Various data suggest an estimated prevalence of ABPM/allergic bronchopulmonary aspergillosis (ABPA) in asthma patients that ranges from 1 to 27.5%.[2],[3] ABPM remains a disease which is less commonly diagnosed or even suspected on first visit to outpatient department (OPD). Most of the cases of ABPA have a history of treatment for pulmonary tuberculosis.[2],[4]

 Case Report 1: Allergic Bronchopulmonary Mycosis and Pulmonary Tuberculosis



A 25-year-old serving soldier, non-smoker, no known co-morbidity was initially evaluated at a peripheral hospital for cough, weight loss, hemoptysis and breathlessness on exertion modified medical research council (MMRC) 0. Investigation: chest X-ray (CXR) posterior anterior (PA) view − non-homogenous opacity left parahilar area [Figure 1], sputum for acid fast bacilli − not seen, sputum for Gram’s stain − cocci seen in chains, haemoglobin − 13.4 g%, total leucocyte counts − 11,000/cumm, differential leucocyte count: neutrophil − 70%, lymphocyte − 21%, eosinophils − 8%, monocyte − 2%, and renal and liver function were normal. He was initially treated for pneumonia, but due to persistence of opacity after 2 weeks of therapy, he was started on anti-tubercular therapy (ATT) and transferred to Military Hospital, Dehradun for supervised daily regimen therapy under pulmonologist. Initial evaluation at Military Hospital, Dehradun revealed bilateral occasional wheeze, and he was continued on ATT. After 15 days of ATT, the patient reported with increased breathlessness on exertion MMRC 2. The patient was re-evaluated and found to have bilateral diffuse wheeze, and CXR PA view revealed opacity in left lower zone [Figure 2] and no evidence of initial CXR findings (opacity in left parahilar finding). The patient was suspected as case of ABPA in view of clinical symptoms and ‘fleeting opacities’. He was further evaluated. Computed tomography (CT) chest revealed a dense area of attenuation of superior segment of left lower lobe and medial segment of medial lobe with central bronchiectasis [Figure 3], serum IgE − 9119.1 IU/L, serum IgE for A. fumigatus − positive, absolute eosinophil counts − 880/cumm, spirometry − moderate obstruction with significant post-bronchodilator reversibility in FEV1 (forced expiratory volume in 1 s change was greater then 200 ml and 12%), sputum for stain and culture yield no bacterial, fungal or tubercular organism. He was diagnosed as of ABPA and started on oral steroid (Tab Prednisolone 50 mg OD, Tab Itraconazole 200 mg OD). ATT was stopped henceforth. The patient has improved significantly with complete resolution of symptoms and now under follow-up.{Figure 1}{Figure 2}{Figure 3}

 Case 2: Allergic Bronchopulmonary Mycosis and Carcinoma Lung



A 62-year-old female, non-smoker, known case of hypertension was initially admitted to a peripheral hospital for cough, streaky hemoptysis and breathlessness on exertion MMRC 1 for 1 month. Investigations: haemoglobin − 13 g%, total leucocyte count − 10,400/cumm, differential leucocyte count: neutrophils − 70%, lymphocytes − 18%, eosinophils − 10%, monocyte − 2%, absolute eosinophil counts − 990/cumm, CXR PA view − mass lesion right hilar area, contrast enhanced CT chest − homogenous opacity superior segment of right lower lobe [Figure 4], sputum examination and culture yield no organism. This patient was referred to Military Hospital, Dehradun as a case of carcinoma lung for further evaluation by a pulmonologist.{Figure 4}

The patient was evaluated in OPD of Military Hospital, Dehradun. Patient had a history of allergy to dust, smoke, cold and recurrent cough. Patient was found to have wheeze on physical examination. CT chest was reviewed and homogenous opacity was seen in superior segment of right lower lobe with surrounding nodular opacities and central bronchiectasis [Figure 4]. She was further evaluated for suspected ABPA in view of history, clinical signs and CT findings. Serum IgE was 14,398.5 IU/L and serum IgE was raised for A. fumigatus, spirometry revealed moderate obstruction and significant post-bronchodilator change in FEV1. Patient was diagnosed a case of ABPA and started on treatment. Patient has shown significant clinical improvement and she is under follow-up.

 Discussion



Aspergillus is the most common fungus involved in ABPM, and pathogenicity in host is determined by host factor. Imbalance between Th1 and Th2 response causing predominant Th2 response to Aspergillus leads to ABPA. Around 25% patient of asthma are sensitised to Aspergillus but only few of them develop ABPA. Prevalence of ABPM is hard to tell due to varied estimation by different studies. But misdiagnosis and wrong treatment to a patient of ABPA are documented well in various case reports and studies. ABPA has been clinically divided into five stages: Stage 1 (acute), Stage 2 (remission), Stage 3 (reoccurrence/exacerbation), Stage 4 (steroid dependent), and Stage 5 (fibrotic lung disease) but patient may not evolve through all stages in sequence.[5] Patient has also been defined in categories of ABPA-central bronchiectasis, ABPA-seropositive, and ABPA-central bronchiectasis with other radiological findings based on radiological and serological investigations.[2] Diagnostic criteria for ABPA are divided into major and minor criteria which includes serology, radiology and sputum examination; hence, combined clinical, serological and radiological evidences are used to diagnose the disease. Rosenberg et al. advised initial criteria for diagnosing ABPA which were later modified by Greenberger et al.

Patient with pulmonary tuberculosis present with chronic cough, fever, weight loss and hemoptysis, and ABPM presents with asthma symptoms with associated fever, weight loss, hemoptysis. Due to similarity in clinical symptoms, endemicity of tuberculosis in India and CXR opacities, it becomes difficult to diagnose or even suspect ABPA on first OPD visit. The first case in our case report was diagnosed as a case of smear negative pulmonary tuberculosis due to non-resolution of CXR opacities. Initial chest findings of wheeze were perhaps neglected due to the history of hemoptysis and the endemicity of tuberculosis in India. Lack of suspicion and awareness is an important cause for missing diagnosis. The patient was suspected as a case of ABPA in the view of breathlessness, with ’fleeting opacities’ and later confirmed with further evaluation. CXR and CT scan may have findings which may get confused with tuberculosis, cryptogenic organising pneumonia, nodular sarcoidosis, chronic eosinophilic pneumonia and some time carcinoma lung in this patient with risk factors such as smoking and advance age. CXR PA view may show ‘tram track’, ‘finger in gloves’, ‘tooth paste shadow’, ‘inverted V or Y’, ‘cluster of grapes’, atelectasis, consolidation, bronchiectasis and nodular lesions. CT scan may show central bronchiectasis (bronchiectasis in medial one third of lung area), mucous plugs, high attenuation mucous − mucous density more then skeletal muscle (due to presence of calcium, iron or haemorrhage in old mucous) and nodular lesions with fibrosis.[6],[7] High attenuation mucous may be present in around 20–30% of cases diagnosed as ABPA.[2],[8]

Difference between bronchiectasis of tuberculosis and ABPA is of different location because it is more distal in tuberculosis whereas it is more proximal in the cases of ABPA causing central bronchiectasis.[9] Central bronchiectasis is a hallmark of disease, and it is present in most of the patients.[10] As the patient was started on ATT without actual disease, a lot of many misdeeds happened. First and foremost is wrong treatment, together with wrong data collection, and more importantly no actual treatment was given to patient, hence, leading to more chances of fibrotic lung disease and disability. ’Soft indication’ for starting ATT as a case of smear negative pulmonary tuberculosis in the case of no sputum positivity in a patient with CXR opacities should not be promoted. One of the reasons for failure of National Tuberculosis Programme was reliability on CXR to diagnose tuberculosis which has wide intra- and inter-observation bias. Review by pulmonologist and further investigation at higher centre may help in curbing wrong treatment.

Second case suspected as a case of carcinoma lung was evaluated for ABPA on first OPD visit due to clinical symptoms, examination finding and CT chest review. Due to mass like appearance of lesion, carcinoma was suspected, perhaps keeping in view of her age. Suspecting carcinoma was a blessing in disguise, because she was not wrongly put on ATT and sent for further evaluation. Both the cases yielded no fungi on sputum examination but sputum positivity varied in various studies from 15.3 to 82.6%.[11] In these case reports, fibre optic bronchoscopy was not done as history, but radiological and serological investigations were good enough to clinch the diagnosis. Serum IgE was raised in both the cases with raised Aspergillus specific IgE, but it may be low in fibrotic lung disease, during remission and patients on oral steroid.[12] Immediate skin reactivity was not done because it was not available. Bronchial asthma has been seen in patients treated for pulmonary tuberculosis which has been attributed to the increase in Th2 response and suppression of Th1 response after treatment with ATT. Most important clue which was missed or ignored was wheeze on initial examination in both cases though endobronchial tuberculosis can mimic as bronchial asthma. Corticosteroids are the main stay of treatment with anti-fungal agents added to decrease antigen load and steroid sparing effect. Human monoclonal anti-IgE antibody (omalizumab) has been used in few case reports, but status regarding its use is still not validated.

 Conclusion



ABPM remains a disease which is still least known, least clinically suspected and a ‘radiological mimicker’ of many illnesses. Awareness of entity, careful history taking and astute clinical examination may give important clinical clue to evaluate further. ‘All wheezes are not asthma’ and ‘Every cough more than 2 weeks should be evaluated for tuberculosis but not treated in haste as tuberculosis’. This becomes more important because most of the cases are treated as a case of pulmonary tuberculosis. Other differential diagnoses such as sarcoidosis, cryptogenic organising pneumonia, chronic eosinophilic pneumonia and carcinoma lung are further evaluated; hence, no wrong treatment is initiated. More awareness may help in curbing wrong diagnosis, wrong treatment and help in adhering to our motto of ‘Primum non nocere’ − first, do no harm.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patients have given their consent for 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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