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Occupational Hypersensitivity Pneumonitis in a BakerHypersensitivity Pneumonitis in a Baker: A New Cause FREE TO VIEW

Mathieu Gerfaud-Valentin, MD; Gabriel Reboux, PhD; Julie Traclet, MD; Françoise Thivolet-Béjui, MD; Jean-François Cordier, MD; Vincent Cottin, MD
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From the Hospices Civils de Lyon (Drs Gerfaud-Valentin, Traclet, Cordier, and Cottin), Hôpital Louis Pradel, Service de Pneumologie-Centre de référence national des maladies pulmonaires rares, Université Claude Bernard Lyon 1, Lyon; Laboratoire de parasitologie et mycologie (Dr Reboux), Centre hospitalier universitaire Jean Minjoz, Besançon; and Hospices Civils de Lyon (Dr Thivolet-Béjui), Groupe hospitalier est, Centre de biologie et pathologie est, Université Claude Bernard Lyon 1, Lyon, France.

Correspondence to: Vincent Cottin, MD, Hospices Civils de Lyon, Hôpital Louis Pradel, Service de Pneumologie-Centre de référence national des maladies pulmonaires rares, Université Claude Bernard Lyon 1, 28 avenue Doyen Lepine, f-69677 Lyon, France; e-mail: vincent.cottin@chu-lyon.fr


Reproduction of this article is prohibited without written permission from the American College of Chest Physicians. See online for more details.


Chest. 2014;145(4):856-858. doi:10.1378/chest.13-1734
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Bakers are exposed daily to flour and may be susceptible to immunologic occupational diseases. A 30-year-old, nonsmoking, female baker was referred for progressive dyspnea on exertion, basal crackles on auscultation, restrictive lung function, decreased diffusing capacity of the lung for carbon monoxide, ground glass hyperdensities with a mosaic pattern on high-resolution CT scan, 25% lymphocytosis by BAL, and cellular chronic bronchiolitis with peribronchiolar interstitial inflammation by lung biopsy specimen. Cultures from flours isolated nine species, including Aspergillus fumigatus. Twenty-six antigens were tested. Serum-specific precipitins were found against A fumigatus, the flour mite Acarus siro, and total extracts from maize and oat. Outcome was favorable with cessation of occupational exposure to flours and transient therapy with prednisone and immunosuppressive agents. To our knowledge, this report is the first of a well-documented case of hypersensitivity pneumonitis due to sensitization to fungi- and mite-contaminated flours. Hypersensitivity pneumonitis—and not only asthma and allergic rhinitis—should be suspected in bakers with respiratory symptoms.

Figures in this Article

Hypersensitivity pneumonitis (HP) is an immunologically mediated diffuse lung disease caused by repeated inhalation of organic agents in sensitized individuals. Causal antigens are largely microbiologic in nature1 and often occupational. Bakers are exposed daily to flour and may develop occupational asthma and atopic rhinitis,2 but, to our knowledge, HP has not been reported previously in bakers.

A 30-year-old female nonsmoker who had been working as a baker for 8 months presented with progressive dyspnea on exertion and dry cough for 3 months. She was exposed daily to wheat and oat flours while making bread from 5:00 pm to 4:00 am and reported morning cough, dyspnea, fever, and chills. She worked in an artisanal family-owned bakery while not wearing a mask in a moderately ventilated room. She had no other relevant history. Symptoms improved while she was away from work during vacations. The patient’s husband also worked in the same bakery but had no symptoms.

Basal inspiratory crackles were present on auscultation. Pulmonary function tests showed the following: total lung capacity, 3.40 L (64% predicted); vital capacity, 2.15 L (61%); FEV1, 2.1 L (60%); diffusing capacity of the lung for carbon monoxide, 33%; and transfer coefficient for carbon monoxide, 60%. Pao2 was 67 mm Hg on room air. Six-minute walk distance was 435 m, with desaturation from 96% to 88%. High-resolution CT (HRCT) scan demonstrated diffuse bilateral ground glass patchy hyperdensities with a mosaic pattern (Fig 1). Laboratory tests showed peripheral blood lymphocytosis (4,510 × 109/L) and moderate polyclonal hypergammaglobulinemia. BAL differential count demonstrated 25% lymphocytes, 5% neutrophils, 3% eosinophils, and 67% macrophages. Antinuclear and anticyclic citrullinated peptide antibodies were absent. A specimen taken from video-assisted lung biopsy of the left lower lobe revealed marked interstitial and bronchiolocentric lymphocytic infiltrates with mild macrophagic alveolitis but without granuloma or fibrosis (Fig 2).

Figure Jump LinkFigure 1. High-resolution CT scan showing diffuse bilateral ground glass patchy hyperdensities with a mosaic pattern. A, Inspiration. B, Expiration.Grahic Jump Location
Figure Jump LinkFigure 2. Lung biopsy specimen demonstrating bronchiolocentric lymphocytic infiltrates (hematoxylin-eosin, magnification × 250).Grahic Jump Location

The patient was referred for further etiologic investigations and management. Microbiologic cultures from 10 flours that the patient used isolated nine species, including Aspergillus fumigatus. Weevils were not found. Twenty-six antigens issued from fungi, actinomycetes, mites, and total flour extracts were tested. Electrosyneresis testing for serum-specific precipitins against A fumigatus and the flour mite Acarus siro were strongly positive, with 4 and 5 arcs, respectively. Moreover, serum testing with total extracts from maize and oat was strongly positive, with 7 and 6 arcs, respectively. IgE specific for Aspergillus was negative.

A diagnosis of HP related to molds and mites contaminating cereal storages and baker’s flours was made. Treatment with 0.5 mg/kg/d po prednisone was initiated with gradual tapering, and the patient was instructed to stop occupational exposure to flours. Clinical symptoms and pulmonary function tests improved initially, but occupational exposure continued, and the patient’s condition deteriorated further. Mycophenolate mofetil was added to corticosteroids with some improvement. The patient eventually avoided persistent exposure to flour, and all drug therapy could then be stopped. Three years later, only functional class 1 dyspnea is present. Mild ground glass hyperdensities are still present on HRCT scan. Pulmonary function tests are as follows: total lung capacity, 3.58 L (71% predicted); vital capacity, 2.0 L (66%); FEV1, 1.54 L (60%); diffusing capacity of the lung for carbon monoxide, 50%; and transfer coefficient for carbon monoxide, 90%.

To our knowledge, this is the first report of a well-documented case of HP due to sensitization to fungi- and mite-contaminated flours in a baker. The diagnosis of HP was based on a combination of features, including a clinical presentation compatible with subacute HP,3 lymphocytic alveolitis on BAL, and a typical imaging pattern with ground glass hyperdensities and mosaic pattern on chest HRCT scan. Histologic confirmation of the diagnosis is not needed in the majority of cases of HP and would not have been mandatory in the present case. The most consistent histologic feature is a cellular chronic bronchiolitis with peribronchiolar interstitial inflammation; poorly formed granulomas (not found in the present case) are present in only two-thirds of cases.4 The diagnosis was further confirmed by strongly positive precipitins against various flours, specifically against molds (Aspergillus) and mites (Acarus) isolated from the flours of the patient’s bakery, and by the dramatic improvement observed after cessation of occupational exposure. A siro, a storage mite, has been shown to be involved in allergic manifestations in millworkers5 and bakers6 yet has not been previously reported as a cause of HP. Aspergillus is a classic contaminant of flours7,8 that may cause HP in other circumstances, especially in farmers.9

Occupational asthma is known to develop in bakers,2 but surprisingly, occupational HP seems to be exceedingly rare, with no case report found in the medical literature through MEDLINE using “hypersensitivity pneumonitis” OR “extrinsic allergic alveolitis” AND “baker” as search terms. Isolated cases of HP due to sensitization to antigens from weevils (beetles that contaminate grains and flours) have been reported10; however, the presence of weevils was definitely ruled out in the present case.

In conclusion, we describe a case of HP due to the inhalation of a variety of flours and their contaminant molds. HP in addition to asthma and allergic rhinitis should be suspected in bakers with respiratory symptoms.

Financial/nonfinancial disclosures: The authors have reported to CHEST that no potential conflicts of interest exist with any companies/organizations whose products or services may be discussed in this article.

Other contributions:CHEST worked with the authors to ensure that the Journal policies on patient consent to report information were met.

HP

hypersensitivity pneumonitis

HRCT

high-resolution CT

Fishwick D. New occupational and environmental causes of asthma and extrinsic allergic alveolitis. Clin Chest Med. 2012;33(4):605-616. [CrossRef]
 
Brant A. Baker’s asthma. Curr Opin Allergy Clin Immunol. 2007;7(2):152-155. [CrossRef]
 
Richerson HB, Bernstein IL, Fink JN, et al; Guidelines for the clinical evaluation of hypersensitivity pneumonitis: report of the Subcommittee on Hypersensitivity Pneumonitis. J Allergy Clin Immunol. 1989;84(5 pt 2):839-844. [CrossRef]
 
Travis WD, Colby TV, Koss MN, Rosado-de-Christenson ML, Mueller NL, Kinh TE. Non-neoplastic disorders of the lower respiratory tract.. In:King DW., ed. American Registry of Pathology and Armed Forces Institute of Pathology. Washington, DC: 2002.
 
Revsbech P, Andersen G. Storage mite allergy among grain elevator workers. Allergy. 1987;42(6):423-429. [CrossRef]
 
Revsbech P, Dueholm M. Storage mite allergy among bakers. Allergy. 1990;45(3):204-208. [CrossRef]
 
Berghofer LK, Hocking AD, Miskelly D, Jansson E. Microbiology of wheat and flour milling in Australia. Int J Food Microbiol. 2003;85(1-2):137-149. [CrossRef]
 
Bosly HA, Kawanna MA. Fungi species and red flour beetle in stored wheat flour under Jazan region conditions [published online ahead of print August 17, 2012]. Toxicol Ind Health. doi:10.1177/0748233712457449.
 
Kaukonen K, Savolainen J, Viander M, Terho EO. Avidity ofAspergillus umbrosusIgG antibodies in farmer’s lung disease. Clin Exp Immunol. 1994;95(1):162-165. [CrossRef]
 
Lunn JA, Hughes DT. Pulmonary hypersensitivity to the grain weevil. Br J Ind Med. 1967;24(2):158-161.
 

Figures

Figure Jump LinkFigure 1. High-resolution CT scan showing diffuse bilateral ground glass patchy hyperdensities with a mosaic pattern. A, Inspiration. B, Expiration.Grahic Jump Location
Figure Jump LinkFigure 2. Lung biopsy specimen demonstrating bronchiolocentric lymphocytic infiltrates (hematoxylin-eosin, magnification × 250).Grahic Jump Location

Tables

References

Fishwick D. New occupational and environmental causes of asthma and extrinsic allergic alveolitis. Clin Chest Med. 2012;33(4):605-616. [CrossRef]
 
Brant A. Baker’s asthma. Curr Opin Allergy Clin Immunol. 2007;7(2):152-155. [CrossRef]
 
Richerson HB, Bernstein IL, Fink JN, et al; Guidelines for the clinical evaluation of hypersensitivity pneumonitis: report of the Subcommittee on Hypersensitivity Pneumonitis. J Allergy Clin Immunol. 1989;84(5 pt 2):839-844. [CrossRef]
 
Travis WD, Colby TV, Koss MN, Rosado-de-Christenson ML, Mueller NL, Kinh TE. Non-neoplastic disorders of the lower respiratory tract.. In:King DW., ed. American Registry of Pathology and Armed Forces Institute of Pathology. Washington, DC: 2002.
 
Revsbech P, Andersen G. Storage mite allergy among grain elevator workers. Allergy. 1987;42(6):423-429. [CrossRef]
 
Revsbech P, Dueholm M. Storage mite allergy among bakers. Allergy. 1990;45(3):204-208. [CrossRef]
 
Berghofer LK, Hocking AD, Miskelly D, Jansson E. Microbiology of wheat and flour milling in Australia. Int J Food Microbiol. 2003;85(1-2):137-149. [CrossRef]
 
Bosly HA, Kawanna MA. Fungi species and red flour beetle in stored wheat flour under Jazan region conditions [published online ahead of print August 17, 2012]. Toxicol Ind Health. doi:10.1177/0748233712457449.
 
Kaukonen K, Savolainen J, Viander M, Terho EO. Avidity ofAspergillus umbrosusIgG antibodies in farmer’s lung disease. Clin Exp Immunol. 1994;95(1):162-165. [CrossRef]
 
Lunn JA, Hughes DT. Pulmonary hypersensitivity to the grain weevil. Br J Ind Med. 1967;24(2):158-161.
 
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