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Cryptogenic organizing pneumonia: A rare extra-intestinal manifestation of Crohn's disease
Journal of Crohn's and Colitis (2014) 8, 177–178
Available online at www.sciencedirect.com
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LETTER TO THE EDITOR Cryptogenic organizing pneumonia: A rare extra-intestinal manifestation of Crohn's disease Dear Sir,
Cryptogenic organizing pneumonia (COP), formerly known as bronchiolitis obliterans organizing pneumonia, is a subacute pulmonary illness characterized by buds of granulation tissue in the bronchioles and alveolar ducts with extension of organization to the alveoli.1,2 It is an exceptionally rare extraintestinal manifestation of Crohn's disease (CD). The shared antigen theory may account for inflammation of both organ systems: decreased IgA secretion from mononuclear cells causes increased permeability to antigens such as bacteria, digestive enzymes and dietary antigens. The common embryonic origin of the GI tract and the bronchial tree from the primitive foregut results in susceptibility of both systems to these antigen.3 We evaluated a 64 year old man with a history of CD maintained on mesalamine for ten years with dyspnea and productive cough for one month. Physical exam was significant for inspiratory crackles on both lung fields. Laboratory data showed leukocytosis with normal eosinophil count. A computed tomography (CT) scan of the chest revealed diffuse ground glass and nodular opacities in bilateral perihilar and lower lung regions (Fig. 1A). Blood, urine, and bronchial cultures were all negative. Mesalamine was held due to concern for drug-induced lung disease. A transbronchial lung biopsy showed elongated “tongues” of loose organizing connective tissue within alveolar ducts typical of COP (Fig. 1B–C). The patient was started on prednisone with complete resolution of symptoms in three weeks. A follow-up CT scan one month after starting prednisone showed almost complete resolution of ground glass opacities in the lungs (Fig. 1D). Mesalamine was restarted without recurrence of pulmonary symptoms. Pulmonary disease in patients with inflammatory bowel disease (IBD) has been described in various forms including tracheobronchitis, bronchiectasis, Langerhans granulomatosis, fibrosing alveolitis, and cryptogenic organizing pneumonia.2 We have found only 4 reported cases of COP in patients with CD and 17 in patients with ulcerative colitis. In these cases, gastrointestinal symptoms and the diagnosis of IBD preceded the onset of pulmonary symptoms, often by several years. COP developed in patients with active as well as inactive IBD. Prior case reports have demonstrated that pulmonary symptoms can develop subacutely following either an inflammatory bowel disease flare or colectomy, or as an independent course.1–4
Determining whether COP is an extra‐intestinal manifestation of CD versus a drug reaction is difficult. Drug-induced lung disease, more commonly presenting as eosinophilic pneumonia, occurs within months of starting the offending agent and typically resolves within weeks of stopping it.2,3,5 In all reported cases in patients taking mesalamine or sulfasalazine there was recurrence of symptoms with a drug re-challenge.5 Our patient tolerated mesalamine for ten years and experienced no recurrence of symptoms upon restarting it making a drug reaction unlikely. COP is treated with systemic corticosteroids and most patients demonstrate a complete recovery.1,2,4,5
References 1. Gil-Simon P, Andres JB, Sanchez RA. Bronchiolitis obliterans organizing pneumonia and Crohn's disease. Rev Esp Enferm Dig 2009;100(3):175–7. 2. Basseri B, Enayati P, Machevsky A. Pulmonary manifestations of inflammatory bowel disease: case presentations and review. J Crohns Colitis 2010;4:390–7. 3. Storch I, Sachar D, Katz S. Pulmonary manifestations of inflammatory bowel disease. Inflamm Bowel Dis 2003;9:104–15. 4. Carratu P, Dragonieri S, Nocerino MC. A case of cryptogenic organizing pneumonia occurring in Crohn's disease. Can Respir J 2005;12(8):437–9. 5. Foster RA, Zander DS, Mergo PJ, Valentine JF. Mesalamine-related lung disease: clinical, radiographic, and pathologic manifestations. Inflamm Bowel Dis 2003;9:308–15.
Henry S. Dinneen Corresponding author: Rutgers New Jersey Medical School 150 Bergen Street, UH-I248 Newark, NJ, USA 07101. E-mail address: [email protected]. Sami Samiullah Rutgers New Jersey Medical School, Newark, NJ, USA Christopher Lenza East Orange Campus of the VA New Jersey Health Care System, East Orange, NJ, USA
1873-9946/$ - see front matter. Published by Elsevier B.V. on behalf of European Crohn's and Colitis Organisation. http://dx.doi.org/10.1016/j.crohns.2013.09.006
6 September 2013
178
Letter to the Editor
Figure 1 A: Initial CT chest showing extensive ground glass and subcentimeter nodular airspace opacities. B: Transbronchial biopsy showing two elongated tongues of organizing connective tissue within alveolar ducts (arrowheads) 40× magnification. C: Organizing tissue within alveolar ducts composed of actively proliferating loose fibroconnective tissue containing scattered inflammatory cells (arrows) 100× magnification. D: Follow-up CT chest showing almost complete resolution of bilateral ground glass opacities.
Available online at www.sciencedirect.com
ScienceDirect
LETTER TO THE EDITOR Cryptogenic organizing pneumonia: A rare extra-intestinal manifestation of Crohn's disease Dear Sir,
Cryptogenic organizing pneumonia (COP), formerly known as bronchiolitis obliterans organizing pneumonia, is a subacute pulmonary illness characterized by buds of granulation tissue in the bronchioles and alveolar ducts with extension of organization to the alveoli.1,2 It is an exceptionally rare extraintestinal manifestation of Crohn's disease (CD). The shared antigen theory may account for inflammation of both organ systems: decreased IgA secretion from mononuclear cells causes increased permeability to antigens such as bacteria, digestive enzymes and dietary antigens. The common embryonic origin of the GI tract and the bronchial tree from the primitive foregut results in susceptibility of both systems to these antigen.3 We evaluated a 64 year old man with a history of CD maintained on mesalamine for ten years with dyspnea and productive cough for one month. Physical exam was significant for inspiratory crackles on both lung fields. Laboratory data showed leukocytosis with normal eosinophil count. A computed tomography (CT) scan of the chest revealed diffuse ground glass and nodular opacities in bilateral perihilar and lower lung regions (Fig. 1A). Blood, urine, and bronchial cultures were all negative. Mesalamine was held due to concern for drug-induced lung disease. A transbronchial lung biopsy showed elongated “tongues” of loose organizing connective tissue within alveolar ducts typical of COP (Fig. 1B–C). The patient was started on prednisone with complete resolution of symptoms in three weeks. A follow-up CT scan one month after starting prednisone showed almost complete resolution of ground glass opacities in the lungs (Fig. 1D). Mesalamine was restarted without recurrence of pulmonary symptoms. Pulmonary disease in patients with inflammatory bowel disease (IBD) has been described in various forms including tracheobronchitis, bronchiectasis, Langerhans granulomatosis, fibrosing alveolitis, and cryptogenic organizing pneumonia.2 We have found only 4 reported cases of COP in patients with CD and 17 in patients with ulcerative colitis. In these cases, gastrointestinal symptoms and the diagnosis of IBD preceded the onset of pulmonary symptoms, often by several years. COP developed in patients with active as well as inactive IBD. Prior case reports have demonstrated that pulmonary symptoms can develop subacutely following either an inflammatory bowel disease flare or colectomy, or as an independent course.1–4
Determining whether COP is an extra‐intestinal manifestation of CD versus a drug reaction is difficult. Drug-induced lung disease, more commonly presenting as eosinophilic pneumonia, occurs within months of starting the offending agent and typically resolves within weeks of stopping it.2,3,5 In all reported cases in patients taking mesalamine or sulfasalazine there was recurrence of symptoms with a drug re-challenge.5 Our patient tolerated mesalamine for ten years and experienced no recurrence of symptoms upon restarting it making a drug reaction unlikely. COP is treated with systemic corticosteroids and most patients demonstrate a complete recovery.1,2,4,5
References 1. Gil-Simon P, Andres JB, Sanchez RA. Bronchiolitis obliterans organizing pneumonia and Crohn's disease. Rev Esp Enferm Dig 2009;100(3):175–7. 2. Basseri B, Enayati P, Machevsky A. Pulmonary manifestations of inflammatory bowel disease: case presentations and review. J Crohns Colitis 2010;4:390–7. 3. Storch I, Sachar D, Katz S. Pulmonary manifestations of inflammatory bowel disease. Inflamm Bowel Dis 2003;9:104–15. 4. Carratu P, Dragonieri S, Nocerino MC. A case of cryptogenic organizing pneumonia occurring in Crohn's disease. Can Respir J 2005;12(8):437–9. 5. Foster RA, Zander DS, Mergo PJ, Valentine JF. Mesalamine-related lung disease: clinical, radiographic, and pathologic manifestations. Inflamm Bowel Dis 2003;9:308–15.
Henry S. Dinneen Corresponding author: Rutgers New Jersey Medical School 150 Bergen Street, UH-I248 Newark, NJ, USA 07101. E-mail address: [email protected]. Sami Samiullah Rutgers New Jersey Medical School, Newark, NJ, USA Christopher Lenza East Orange Campus of the VA New Jersey Health Care System, East Orange, NJ, USA
1873-9946/$ - see front matter. Published by Elsevier B.V. on behalf of European Crohn's and Colitis Organisation. http://dx.doi.org/10.1016/j.crohns.2013.09.006
6 September 2013
178
Letter to the Editor
Figure 1 A: Initial CT chest showing extensive ground glass and subcentimeter nodular airspace opacities. B: Transbronchial biopsy showing two elongated tongues of organizing connective tissue within alveolar ducts (arrowheads) 40× magnification. C: Organizing tissue within alveolar ducts composed of actively proliferating loose fibroconnective tissue containing scattered inflammatory cells (arrows) 100× magnification. D: Follow-up CT chest showing almost complete resolution of bilateral ground glass opacities.