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Changing Pulmonary Infiltrates in ABPA Misdiagnosed As Recurrent Pneumonia: a Case Report and Review of the Literature

Changing Pulmonary Infiltrates in ABPA Misdiagnosed As Recurrent Pneumonia: a Case Report and Review of the Literature

Respir Case Rep 2018;7(3):134-139 DOI: 10.5505/respircase.2018.20082

OLGU SUNUMU CASE REPORT

Changing Pulmonary Infiltrates in ABPA Misdiagnosed as Recurrent : A Case Report and Review of the Literature

Tekrarlayan Pnömoni Olarak Yanlış Tanı Konan ABPA Olgusunda Değişen Pulmoner İnfiltratlar: Olgu Sunumu ve Literatürün Gözden Geçirilmesi

Shital Patil, Gajanan Gondhali

Abstract Özet Pneumonia is the most common respiratory problem Pnömoni, Hindistan gibi tropikal ülkelerde en sık in tropical countries like India. Allergic bronchopul- görülen solunumsal problemdir. Allerjik bronkopul-

monary aspergillosis (ABPA) is routinely underdiag- moner aspergillozis (ABPA), pnömoni ve tüberküloz

nosed and under evaluated as a result of clinical and gibi solunumsal hastalıklar ile klinik ve radyolojik radiological overlap with many other respiratory olarak karıştırıldığı için genellikle tanıda ve inceleme- conditions, including pneumonia and . lerde atlanmaktadır. ABPA, uzun süreli atopik astmalı ABPA is the best-recognized manifestation of the hastalarda Aspergillus antijenine aşırı duyarlılık ile Aspergillus, associated with hypersensitivity to karakterize, aspergillus mantarına ait en iyi tanım- Aspergillus in patients with longstanding lanmış bir tablodur. ABPA’nın hastaneye yatırılan atopic . ABPA has been reported to occur in astmatik hastaların %20’sinde ve tüm rinit olgularının 20% of asthmatic patients admitted to hospitals and %5’inde çeşitli klinik tablolarla ortaya çıktığı bildiril- in 5% of all cases, with varied clinical presen- miştir. Burada, öksürük, ateş, nefes darlığı gibi ya- tations. This report is a description of the case of a kınmaları ile tekrarlayan pnömoni tanısı ile tedavi middle-aged male with known asthma for several edilen, birkaç yıldır astım olduğu bilinen orta yaşlı years and constitutional symptoms, such as a , erkek olgu sunulmuştur. Bu olguda, sonuçta ABPA , and , who was diagnosed olarak tanı konmuş ve sistemik steroid ve antifungal and treated for recurrent pneumonia. The case was tedavi ile tam klinik ve radyolojik iyileşme sağlanmış- eventually confirmed as ABPA, and a complete clini- tır. cal and radiological response to medical treatment Anahtar Sözcükler: ABPA, tekrarlayan pnömoni, pul- with antifungals and systemic was moner infiltrat. documented. Key words: ABPA, Recurrent pneumonia, fleeting pulmonary infiltrates.

RESPIRATORY CASE REPORTS CASE RESPIRATORY Allergic bronchopulmonary aspergillosis (ABPA), response (2). ABPA is still under-recognized and the most widely studied Aspergillus-related allergic underdiagnosed in India, in spite of its relatively phenomenon, is an immune-mediated inflamma- high prevalence. Many factors may contribute to tory syndrome caused by hypersensitivity to a ubiq- this situation. However, the clinical presentation of uitous fungus, Aspergillus fumigatus (1). The clini- ABPA may be indistinguishable from pneumonia or cal, radiological, and histological manifestations pulmonary tuberculosis, especially in developing of bronchopulmonary aspergillosis depend not countries where the prevalence of pulmonary tu- only on the number and of the infective berculosis is still high (3). organism, but also on the patient’s immune

MIMSR Medical College, Latur, India MIMSR Tıp Fakültesi, Latur, India Submitted (Başvuru tarihi): 13.02.2018 Accepted (Kabul tarihi): 06.04.2018

Correspondence (İletişim): Shital Patil, MIMSR Medical College, Latur, India e-mail: [email protected]

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CASE evaluation- bilateral and A 44-year-old man, with known bronchial asthma for 15 coarse years, presented with a cough with minimal pro- Ear/nose/throat evaluation- bilateral nasal turbinate hy- duction, shortness of breath (Grade IV), and a low-grade pertrophy intermittent fever for 15 days. Test results: He had been using inhaled bronchodilators and inhaled Hemoglobin-13.8 gm% corticosteroids as treatment for 15 years, with Total count- 16000/mm3. a history of infrequent exacerbation and hospitalization Eosinophil percentage- 9% (of total differential cell count) for the same conditions. Absolute eosinophil count- 1276/mm3 An X-ray done at a general hospital indicated heteroge- Sputum eosinophil count- 8% neous opacity filling the left upper zone air space (Figure Sputum for acid-fast bacilli- negative with Zeihl-Neelsen 1). He was diagnosed with community-acquired pneu- stain monia and empirical treatment with Sputum for GeneXpert Mycobacterium tuberculo- and was initiated. The clinical response to sis/rifampicin (MTB/RIF) (Cepheid, Inc., Sunnyvale, CA, and bronchodilators was not satisfactory, his USA)- negative for MTB genome breathlessness worsened, and the patient was referred to for Mycobacterium tuberculosis-negative the pulmonary medicine department after 4 weeks in liquid media (Middlebrook 7H9 media) for respiratory care and further expert management. Serum immunoglobulin E (IgE) level - 1036 ng/mL The patient reported experiencing similar episodes 1 year Serum precipitins- positive serum (precipitins) earlier that were diagnosed as community-acquired for Aspergillus species pneumonia and treated with empirical antibiotics; howev- Sputum culture for fungus- Aspergillus fumigatus identi- er the treatment provided only partial relief of symptoms. fied in fungal culture Underlying bronchial asthma may lead to misleading Final diagnosis- confirmed as ABPA symptoms and an incorrect diagnosis. This case was confirmed as ABPA and the patient was started on antifungals and steroids: itraconazole and omnacortil. We documented radiological and clinical response in the first month (Figure 2), which is rare in tuberculosis where radiological response is often delayed.

Figure 1: Showing left upper zone air space heterogeneous opacity

Examination findings:

Respiratory rate- 24/per minute, functional accessory Figure 2: Response to treatment after one month muscles of respiration Partial saturation- 90% room air (improved to This patient received itraconazole at a dose of 100 mg 96% at nasal oxygen 3 L/minute) twice daily for 16 weeks and omnacortil at dose of 2 rate – 102/minute mg/kg, which was tapered gradually over 24 weeks. - 100/60 MmHg

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Near total resolution of all radiological shadows was A high-resolution computed tomography image of the observed after 3 months of treatment (Figure 3). thorax documented changing (fleeting) pulmonary infil- The classic sign of fleeting pulmonary infiltrates, as well trates involving the right lower lobe (Figure 6) and left as other radiological signs were observed (Figures 4-9). upper lobe (Figure 7). A high-resolution computed tomography image of the A high-resolution computed tomography of the thorax thorax showing air space consolidation with air- documented the finger-in-glove sign (Figure 8) and cen- bronchogram involving the right posterior segment (Fig- tral (Figure 9). ure 4), bilateral centrilobular nodules and lingular seg- ment (Figure 5).

Figure 6: Right lower lobe

Figure 3: Response to treatment after 3 months

Figure 7: Left upper lobe

DISCUSSION ABPA was first described in 1952 when Hinson, Moon, and Plummer wrote of 3 patients with recurrent wheezing, pulmonary infiltrates, eosinophilia in the blood and spu- tum, and brown plugs or flecks in expectorated mucus. Figure 4: Right posterior segment Clinically, ABPA presents like increasingly severe asthma

or exacerbation of cystic (CF) (4). There are no specific clinical or findings. The symptoms can range from recurrent pulmonary exacerba- tions with cough, wheezing, and shortness of breath, to systemic features, including fever, , and . Physical examination findings can range from normal results to the observation of digital clubbing, auscultatory fine crackles, or bronchial breath sounds (4). Two differential diagnoses for ABPA include and pulmonary tuberculosis, which should be

Figure 5: Lingular segment

136 www.respircase.com Respiratory Case Reports given great care in India and other areas where there is a Modified International Society for Human and Animal high prevalence (3). Mycology Working Group 2013 criteria for diagnosis of It was not until 1977 that Rosenberg, Patterson et al. (5) ABPA (6) proposed a set of diagnostic criteria. 1. Predisposing asthma or 2. Obligatory criteria a. IgE >1000 IU/mL and b. Positive immediate skin test or elevated level of IgE antibodies to Aspergillus 3. Supportive (>2) criteria a. Eosinophilia >500 b. Precipitins or increased IgG antibodies to Aspergillus c. Consistent radiographic opacities Mendelson et al. (7) described chest radiographic find- ings in various stages of ABPA.

Figure 8: Finger in glove appearance Stage Description Radiological findings I phase Normal, pulmonary infiltrates and mucoid impaction, predominantly in the upper lobes

II Remission Significant resolution of pulmonary infil- trates and clearance of mucoid impaction

III Exacerbation Reappearance of infiltrates and/or mu- coid impaction in previously involved, as well as new, areas

IV Glucocorticoid Significant resolution of pulmonary infil- dependent trates and mucoid impaction, although ABPA fixed pulmonary opacities may be en- countered Figure 9: Central bronchiectasis V End-stage Evidence of bronchiectasis, pulmonary (fibrotic) ABPA fibrosis, pulmonary Primary criteria (1–6 suggestive, +7 definite) 1. Episodic bronchial obstruction ABPA: Allergic bronchopulmonary aspergillosis 2. Peripheral eosinophilia 3. Positive immediate skin test to Aspergillus The active stage is characterized radiographically by tran- 4. Positive precipitin test to Aspergillus sient and recurrent infiltrates that may clear with or with- 5. Increased total serum IgE out glucocorticoid therapy, although steroid therapy does 6. History of transient or fixed infiltrates hasten the clearing of opacities. Consolidation is believed 7. Proximal bronchiectasis to be one of the most common findings, and the occur- rence of has also been patho- Secondary (supportive) criteria logically demonstrated (8). 1. Brown plugs/flecks in sputum Tram-line shadows, band-like (toothpaste) shadows show- 2. Positive late (6–12 h/Arthus) skin test to Aspergillus ing sometimes V-shaped, inverted V-, or Y-shaped shad- ows, and finger-in-glove opacities may occur. These are Since then, as laboratory and clinical medicine have the most characteristic finding of ABPA and represent continued to advance, diagnostic criteria for ABPA have mucoid impaction in dilated bronchi with occlusion of the been modified, especially in light of improved and more distal end. These shadows are often transient, disappear- specific serological and radiographic testing (6). ing with the expulsion of secretions either spontaneously or following treatment (8). Central bronchiectasis (CB) is believed to be a characteristic finding in ABPA, although

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there are no uniform criteria for the diagnosis of CB. tients met immunological and pulmonary function test Depending on the proximity of the dilated bronchi from criteria for -dependent ABPA. the hilum at a point midway between the hilum and the A response was defined as at least a 50% reduction in chest wall, bronchiectasis is defined as central if confined steroid dose, a 25% reduction in serum IgE concentration, to the medial two-thirds or the medial half of the lung (1). and either an improvement of 25% in exercise tolerance Bronchiectasis can, however, extend to the periphery as testing or pulmonary function testing or a resolution of well, and peripheral bronchiectasis has been described in pulmonary infiltrates on imaging. There was also a fol- 26% to 39% of lobes affected by bronchiectasis. The low-on open-label arm of the trial where all of the pa- bronchiectasis in ABPA usually involves the upper lobes, tients received itraconazole 200 mg daily (a lower dose although rarely, there may be involvement of the lower than in the placebo-controlled trial) for 16 weeks. The zones without involvement of the upper lobes (1). study demonstrated that in patients with corticosteroid- Systemic steroids have been shown to be an effective first- dependent ABPA, adding itraconazole can lead to clinical line treatment for APBA in both asthma and CF. Agarwal improvement without significant risk of toxicity. Addition- et al. (9) described a more aggressive approach with a ally, the lower dose used in the open-label trial showed a treatment dose of 0.75 mg/kg/day for 6 weeks, then 0.5 benefit as well (12). mg/kg/day for 6 weeks, followed by a tapering dose of 5 mg every 6 weeks for a total duration of 6 to 12 months. CONCLUSION Recently, Agarwal et al. (10) performed a randomized ABPA has a diverse clinical presentation, ranging from controlled trial with patients who had a diagnosis of typical bronchial asthma to tropical infectious pulmonary asthma and ABPA comparing the efficacy and safety of diseases, like pneumonia and tuberculosis. All possible the 2 regimens. The 0.5 mg/kg/day regimen was referred measures should be taken to rule out ABPA, especially in to as the “medium dose,” while the 0.75 mg/kg/day a scenario with recurrent pneumonia, for example, with regimen was referred to as a “high dose” regimen. Previ- changing pulmonary infiltrates. ous studies had looked at each regimen individually and A high index of suspicion is necessary while managing there was some suggestion that the high dose would be these cases, particularly in tropical countries, like India, superior in the prevention of exacerbation (9). This study where pneumonia is a frequently encountered respiratory was the first randomized, controlled trial to compare 2 issue. ABPA is easily managed with antifungals and ster- steroid regimens, and it was determined that the medium oids if diagnosed early, and the treatment outcome will dose of oral glucocorticosteroids (prednisolone) was both likely be successful if diagnosed before it reaches the effective and safer than the high dose in the treatment of fibrosis stage. ABPA (10). Adding an antifungal agent to the regimen may have a CONFLICTS OF INTEREST steroid-sparing effect, reducing the need for steroids to None declared. control (11). Azoles are used to reduce the burden arising from fungal colonization of the AUTHOR CONTRIBUTIONS airway. It is then expected that the reduction in antigenic Concept - S.P., G.G.; Planning and Design - S.P., G.G.; stimulation would result in decreased inflammation and Supervision - S.P., G.G.; Funding - S.P.; Materials - S.P.; reduced disease severity and progression. Itraconazole is Data Collection and/or Processing - G.G.; Analysis an orally administered triazole that has fewer side effects and/or Interpretation - G.G.; Literature Review - S.P.; and a wider spectrum of activity compared with ketocon- Writing - S.P.; Critical Review - S.P. azole. There have been open-label case series that sug- gest benefit in the treatment of ABPA in patients with and YAZAR KATKILARI without CF (11). Fikir - S.P., G.G.; Tasarım ve Dizayn - S.P., G.G.; Denet- There are 2 randomized controlled trials in the literature leme - S.P., G.G.; Kaynaklar - S.P.; Malzemeler - S.P.; using itraconazole in ABPA. Stevens et al. (12) in 2000 Veri Toplama ve/veya İşleme - G.G.; Analiz ve/veya published findings from their randomized, double-blind Yorum - G.G.; Literatür Taraması - S.P.; Yazıyı Yazan - trial of treatment with either 200 mg of itraconazole twice S.P.; Eleştirel İnceleme - S.P. daily or a placebo for 16 weeks in patients. These pa-

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