A case of allergic bronchopulmonary aspergillosis in a 43-year-old farmer following single high level exposure to organic dust with symptomatic remission and radiological resolution after early diagnosis and treatment

A case of allergic bronchopulmonary aspergillosis in a 43-year-old farmer following single high... Allergic bronchopulmonary aspergillosis (ABPA) is an uncommon lung condition associated with development or worsening of asthma symptoms, distinctive radiological and serological findings. It is thought to be the consequence of chronic colonization of the airways in individuals with pre-existing atopic conditions. We present a case unique in the literature of the development of ABPA in an individual without pre-existing atopic disease following a single high level exposure to organic dust. Early treatment was associated with complete symptomatic remission and significant resolution of bronchiectatic changes. Here we report a case of a patient without pre-existing asth- INTRODUCTION ma or atopic disease developing ABPA after a single exposure Allergic bronchopulmonary aspergillosis (ABPA) is an uncom- to high concentration of fungal spores. Treatment including mon lung condition associated with an allergic immunological steroid and antifungal therapy was associated with complete response to airway Aspergillus fumigatus. It is normally seen in resolution of asthma symptoms allowing treatment withdrawal those with pre-existing asthma or cystic fibrosis occasionally and with partial resolution of bronchiectatic changes. other atopic disease [1]. It is associated with radiological changes including focal infiltrates, segmental lung collapse and bronchiectasis, microbiological and serological evidence CASE REPORT of Aspergillus. These together form criteria for the diagnosis of this condition [2]. Little is known of the natural history of A 43-year-old farmer presented to outpatients with a 3-week ABPA but, whilst infiltrates and collapse are associated with history of wheeze, shortness of breath and cough productive of eosinophillic inflammation and are reversible changes, bron- firm rubbery mucous plugs. His symptoms started following a chiectasis and asthma, once established, are thought to be single exposure to clouds of dust produced during the disposal irreversible [3]. of piles of rotting hedge clippings. The cough and wheeze were Received: June 21, 2017. Revised: November 8, 2017. Accepted: November 23, 2017 © The Author 2018. Published by Oxford University Press. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/omcr/article-abstract/2018/2/omx091/4846367 by Ed 'DeepDyve' Gillespie user on 16 March 2018 A case of allergic bronchopulmonary aspergillosis in a 43-year-old farmer 43 (a) (b) Mucous-filled bronchi Centri-lobular nodules Figure 1: (a) CXR showing ‘gloved finger’ change (arrow). (b) CT thorax showing centri-lobular nodules and mucous-filled bronchi corresponding to CXR changes worst at night and in the morning. There was no previous his- tory of respiratory or atopic disease. On examination there was widespread audible expiratory wheeze. His peak expiratory flow was 400 L/min (63% pre- dicted). CXR showed bilateral upper zone infiltrates with left upper zone changes consistent with the ‘gloved finger’ change (Fig. 1). CT thorax showed findings consistent with the diagnosis of ABPA: bilateral central bronchiectatic changes with multiple mucous-filled airways in the upper lobes corresponding to the CXR abnormality and associated with peripheral centri-lobular nodules (Fig. 1). Blood tests showed high total IgE levels 1454 kU/L (ULN 81.0), strongly positive A. fumigatus-specific IgE 40.5 kU/L (ULN < 9 9 0.35) and elevated eosinophil count 1.1 × 10 /L (ULN 0.4 × 10 /L), Figure 2: Time course of Aspergillus fumigatus-specific IgE titre before, during Aspergillus precipitins were negative but sputum culture grew and after treatment A. fumigatus and cytological examination of bronchoalveolar lavage fluid showed branching fungal hyphae. specific antibodies and in the context of his illness the diagno- Based on diagnostic criteria the diagnosis of ABPA was con- sis of ABPA seems secure. He described no symptoms suggest- firmed and treatment started with oral Prednisolone 30 mg and ing pre-existing atopic lung disease and his Primary Care Itraconazole 200 mg bd. His symptoms resolved rapidly with record did not suggest undiagnosed disease for example there improvement in lung function PEF 700 L/min (111% predicted) was no history of recurrent chest infection or previous elevated FEV1 4.5 L/s (111% predicted) and clearance of CXR changes. blood eosinophil count. Aspergillus serology showed progressive improvement Chronic airway colonization with aspergillus is thought (Fig. 2) and total IgE decreased substantially to 333 kU/L. to create an immunological response leading to ABPA [1]. After 12 months of therapy Prednisolone, Itraconazole Aspergillus is ubiquitous in the environment and index expos- 200 mg bd and inhaled corticosteroid therapy were stopped ure is rarely identified. There are occasional case reports iden- with no deterioration in symptoms or lung function. tifying a potential source of chronic exposure [4–6] but this is A follow-up CT thorax at 30 months showed complete reso- the first reported case of a single high level exposure leading lution of the centri-lobular nodules, infiltrates and mucous to abrupt onset of symptoms. plugging with partial resolution of bronchiectatic changes Exposure to sensitizing allergens is known to cause asthma (Fig. 3). The bronchiectasis persisted in the LUL bronchi most in those with no pre-existing atopic disease [7] but this is the affected by mucous plugging. first report of ABPA arising in an individual with no history of pre-existing atopy, asthma or cystic fibrosis. DISCUSSION Little is known of the natural history of ABPA but consistent The diagnosis of ABPA is based on a number of criteria includ- with other conditions associated with asthma and with bron- chiectasis, ABPA is thought of as a chronic disease with a ing asthma, total IgE > 412 IU/l, elevated Aspergillus species specific IgE and IgG, CXR infiltrates, central bronchiectasis and relapsing remitting course and no cure [8]. Although treatment Aspergillus cultured from sputum and/or fungal hyphae seen with oral steroids and Itraconazole improves symptoms and in mucous plugs [2]. Our patient met all criteria barring IgG slows disease progression in many [9], once bronchiectatic or Downloaded from https://academic.oup.com/omcr/article-abstract/2018/2/omx091/4846367 by Ed 'DeepDyve' Gillespie user on 16 March 2018 44 P. Flood-Page 2014 2017 Mucous filled dilated bronchus Centri-lobular nodules Figure 3: CT thorax of lower lobe changes before (2014) and after (2017) treatment showing resolution of centri-lobular nodules and mucous impaction/ bronchiectasis fibrotic changes are established, they are not thought to be GUARANTOR reversible. Here we present for the first time evidence that early P.F.P. is the guarantor of this article. diagnosis and intervention is associated with complete symp- tomatic remission of asthma with reversal of bronchiectatic change. Although Aspergillus-specific serological tests are REFERENCES markedly diminished and continue to fall, they are still ele- 1. Douglas Robinson and Patrick Flood-Page. Bronchopulmonary vated, some bronchiectatic change persists and the potential aspergillosis. In: Respiratory Medicine, 3rd edn. Chapter 40 for future relapse remains. Our patient, however, remains Bronchopulmonary Aspergillosis, pp. 986–1010, Saunder symptom-free over 12 months post treatment cessation and Press. complete cure with normalization of serology and continued 2. Knutsen AP, Bush RK, Demain JG, Denning DW, Dixit A, Fairs resolution of bronchiectasis is possible. A, et al. Fungal and allergic lower respiratory tract diseases. J Allergy Clin Immunol 2012;129:280–91. CONCLUSION 3. McCarthy DS, Simon G, Hargreave FE. The radiological This case reports a number of new findings in ABPA. It is the first appearances in allergic broncho-pulmonary aspergillosis. reported case of ABPA associated with a single high level expos- Clin Radiology 1970;21:366–75. ure to A. fumigatus.Itisthe first report of ABPA occurring in an 4. Kramer MN, Kurup VP, Fink JN. Allergic bronchopulmonary individual with no prior history of atopic disease. It is the first aspergillosis from a contaminated dump site. Am Rev Respir reported instance of complete and sustained symptomatic reso- Dis 1989;140:1086–8. lution, partial resolution of bronchiectatic change and potential 5. Poole CJ, Wong M. Allergic bronchopulmonary aspergillosis ABPA cure. It highlights the potential importance of early diagno- in garden waste (compost) collectors: occupational implica- sis and treatment to the natural history of this condition. tions. Occup Med (Lond) 2013;63:517–9. 6. Allmers H, Huber H, Baur X. Two year follow-up of garbage collector with allergic bronchopulmonary aspergillosis (ABPA). CONFLICT OF INTEREST STATEMENT Am J Med 2000;37:438–42. There is no conflict of interest. 7. Peden D, Reed CE. Environmental and occupational allergies. J Allergy Clin Immunol 2010;125(2 Suppl 2):S150–60. FUNDING 8. Greenberger PA. Allergic bronchopulmonary aspergillosis. J Allergy Clin Immunol 2002 Nov;110:685–92. There is no funding source. 9. Stevens DA, Schwartz HJ, Lee JY, Moskovitz BL, Jerome DC, Catanzaro A, et al. A randomized trial of itraconazole in aller- ETHICAL APPROVAL AND CONSENT gic bronchopulmonary aspergillosis. N Engl J Med 2000;342: Patient consent was obtained. 756–62. Downloaded from https://academic.oup.com/omcr/article-abstract/2018/2/omx091/4846367 by Ed 'DeepDyve' Gillespie user on 16 March 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Oxford Medical Case Reports Oxford University Press

A case of allergic bronchopulmonary aspergillosis in a 43-year-old farmer following single high level exposure to organic dust with symptomatic remission and radiological resolution after early diagnosis and treatment

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Abstract

Allergic bronchopulmonary aspergillosis (ABPA) is an uncommon lung condition associated with development or worsening of asthma symptoms, distinctive radiological and serological findings. It is thought to be the consequence of chronic colonization of the airways in individuals with pre-existing atopic conditions. We present a case unique in the literature of the development of ABPA in an individual without pre-existing atopic disease following a single high level exposure to organic dust. Early treatment was associated with complete symptomatic remission and significant resolution of bronchiectatic changes. Here we report a case of a patient without pre-existing asth- INTRODUCTION ma or atopic disease developing ABPA after a single exposure Allergic bronchopulmonary aspergillosis (ABPA) is an uncom- to high concentration of fungal spores. Treatment including mon lung condition associated with an allergic immunological steroid and antifungal therapy was associated with complete response to airway Aspergillus fumigatus. It is normally seen in resolution of asthma symptoms allowing treatment withdrawal those with pre-existing asthma or cystic fibrosis occasionally and with partial resolution of bronchiectatic changes. other atopic disease [1]. It is associated with radiological changes including focal infiltrates, segmental lung collapse and bronchiectasis, microbiological and serological evidence CASE REPORT of Aspergillus. These together form criteria for the diagnosis of this condition [2]. Little is known of the natural history of A 43-year-old farmer presented to outpatients with a 3-week ABPA but, whilst infiltrates and collapse are associated with history of wheeze, shortness of breath and cough productive of eosinophillic inflammation and are reversible changes, bron- firm rubbery mucous plugs. His symptoms started following a chiectasis and asthma, once established, are thought to be single exposure to clouds of dust produced during the disposal irreversible [3]. of piles of rotting hedge clippings. The cough and wheeze were Received: June 21, 2017. Revised: November 8, 2017. Accepted: November 23, 2017 © The Author 2018. Published by Oxford University Press. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/omcr/article-abstract/2018/2/omx091/4846367 by Ed 'DeepDyve' Gillespie user on 16 March 2018 A case of allergic bronchopulmonary aspergillosis in a 43-year-old farmer 43 (a) (b) Mucous-filled bronchi Centri-lobular nodules Figure 1: (a) CXR showing ‘gloved finger’ change (arrow). (b) CT thorax showing centri-lobular nodules and mucous-filled bronchi corresponding to CXR changes worst at night and in the morning. There was no previous his- tory of respiratory or atopic disease. On examination there was widespread audible expiratory wheeze. His peak expiratory flow was 400 L/min (63% pre- dicted). CXR showed bilateral upper zone infiltrates with left upper zone changes consistent with the ‘gloved finger’ change (Fig. 1). CT thorax showed findings consistent with the diagnosis of ABPA: bilateral central bronchiectatic changes with multiple mucous-filled airways in the upper lobes corresponding to the CXR abnormality and associated with peripheral centri-lobular nodules (Fig. 1). Blood tests showed high total IgE levels 1454 kU/L (ULN 81.0), strongly positive A. fumigatus-specific IgE 40.5 kU/L (ULN < 9 9 0.35) and elevated eosinophil count 1.1 × 10 /L (ULN 0.4 × 10 /L), Figure 2: Time course of Aspergillus fumigatus-specific IgE titre before, during Aspergillus precipitins were negative but sputum culture grew and after treatment A. fumigatus and cytological examination of bronchoalveolar lavage fluid showed branching fungal hyphae. specific antibodies and in the context of his illness the diagno- Based on diagnostic criteria the diagnosis of ABPA was con- sis of ABPA seems secure. He described no symptoms suggest- firmed and treatment started with oral Prednisolone 30 mg and ing pre-existing atopic lung disease and his Primary Care Itraconazole 200 mg bd. His symptoms resolved rapidly with record did not suggest undiagnosed disease for example there improvement in lung function PEF 700 L/min (111% predicted) was no history of recurrent chest infection or previous elevated FEV1 4.5 L/s (111% predicted) and clearance of CXR changes. blood eosinophil count. Aspergillus serology showed progressive improvement Chronic airway colonization with aspergillus is thought (Fig. 2) and total IgE decreased substantially to 333 kU/L. to create an immunological response leading to ABPA [1]. After 12 months of therapy Prednisolone, Itraconazole Aspergillus is ubiquitous in the environment and index expos- 200 mg bd and inhaled corticosteroid therapy were stopped ure is rarely identified. There are occasional case reports iden- with no deterioration in symptoms or lung function. tifying a potential source of chronic exposure [4–6] but this is A follow-up CT thorax at 30 months showed complete reso- the first reported case of a single high level exposure leading lution of the centri-lobular nodules, infiltrates and mucous to abrupt onset of symptoms. plugging with partial resolution of bronchiectatic changes Exposure to sensitizing allergens is known to cause asthma (Fig. 3). The bronchiectasis persisted in the LUL bronchi most in those with no pre-existing atopic disease [7] but this is the affected by mucous plugging. first report of ABPA arising in an individual with no history of pre-existing atopy, asthma or cystic fibrosis. DISCUSSION Little is known of the natural history of ABPA but consistent The diagnosis of ABPA is based on a number of criteria includ- with other conditions associated with asthma and with bron- chiectasis, ABPA is thought of as a chronic disease with a ing asthma, total IgE > 412 IU/l, elevated Aspergillus species specific IgE and IgG, CXR infiltrates, central bronchiectasis and relapsing remitting course and no cure [8]. Although treatment Aspergillus cultured from sputum and/or fungal hyphae seen with oral steroids and Itraconazole improves symptoms and in mucous plugs [2]. Our patient met all criteria barring IgG slows disease progression in many [9], once bronchiectatic or Downloaded from https://academic.oup.com/omcr/article-abstract/2018/2/omx091/4846367 by Ed 'DeepDyve' Gillespie user on 16 March 2018 44 P. Flood-Page 2014 2017 Mucous filled dilated bronchus Centri-lobular nodules Figure 3: CT thorax of lower lobe changes before (2014) and after (2017) treatment showing resolution of centri-lobular nodules and mucous impaction/ bronchiectasis fibrotic changes are established, they are not thought to be GUARANTOR reversible. Here we present for the first time evidence that early P.F.P. is the guarantor of this article. diagnosis and intervention is associated with complete symp- tomatic remission of asthma with reversal of bronchiectatic change. Although Aspergillus-specific serological tests are REFERENCES markedly diminished and continue to fall, they are still ele- 1. Douglas Robinson and Patrick Flood-Page. Bronchopulmonary vated, some bronchiectatic change persists and the potential aspergillosis. In: Respiratory Medicine, 3rd edn. Chapter 40 for future relapse remains. Our patient, however, remains Bronchopulmonary Aspergillosis, pp. 986–1010, Saunder symptom-free over 12 months post treatment cessation and Press. complete cure with normalization of serology and continued 2. Knutsen AP, Bush RK, Demain JG, Denning DW, Dixit A, Fairs resolution of bronchiectasis is possible. A, et al. Fungal and allergic lower respiratory tract diseases. J Allergy Clin Immunol 2012;129:280–91. CONCLUSION 3. McCarthy DS, Simon G, Hargreave FE. The radiological This case reports a number of new findings in ABPA. It is the first appearances in allergic broncho-pulmonary aspergillosis. reported case of ABPA associated with a single high level expos- Clin Radiology 1970;21:366–75. ure to A. fumigatus.Itisthe first report of ABPA occurring in an 4. Kramer MN, Kurup VP, Fink JN. Allergic bronchopulmonary individual with no prior history of atopic disease. It is the first aspergillosis from a contaminated dump site. Am Rev Respir reported instance of complete and sustained symptomatic reso- Dis 1989;140:1086–8. lution, partial resolution of bronchiectatic change and potential 5. Poole CJ, Wong M. Allergic bronchopulmonary aspergillosis ABPA cure. It highlights the potential importance of early diagno- in garden waste (compost) collectors: occupational implica- sis and treatment to the natural history of this condition. tions. Occup Med (Lond) 2013;63:517–9. 6. Allmers H, Huber H, Baur X. Two year follow-up of garbage collector with allergic bronchopulmonary aspergillosis (ABPA). CONFLICT OF INTEREST STATEMENT Am J Med 2000;37:438–42. There is no conflict of interest. 7. Peden D, Reed CE. Environmental and occupational allergies. J Allergy Clin Immunol 2010;125(2 Suppl 2):S150–60. FUNDING 8. Greenberger PA. Allergic bronchopulmonary aspergillosis. J Allergy Clin Immunol 2002 Nov;110:685–92. There is no funding source. 9. Stevens DA, Schwartz HJ, Lee JY, Moskovitz BL, Jerome DC, Catanzaro A, et al. A randomized trial of itraconazole in aller- ETHICAL APPROVAL AND CONSENT gic bronchopulmonary aspergillosis. N Engl J Med 2000;342: Patient consent was obtained. 756–62. Downloaded from https://academic.oup.com/omcr/article-abstract/2018/2/omx091/4846367 by Ed 'DeepDyve' Gillespie user on 16 March 2018

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Published: Feb 1, 2018

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