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Mucoid Plugs in Eosinophilic Pneumonia

Cumbo-Nacheli, Gustavo MD; Ashton, Rendell W. MD

Journal of Bronchology & Interventional Pulmonology: July 2011 - Volume 18 - Issue 3 - p 257–258
doi: 10.1097/LBR.0b013e318222a043
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A 47-year-old woman developed dyspnea, sputum production, and intermittent cough, which persisted for several months. She had received treatment for asthma without resolution of symptoms. Radiologic evidence of bilateral pulmonary infiltrates prompted a bronchoscopic examination, which showed extensive bronchial plugging. The material had a mucoid appearance. Bronchioalveolar lavage showed abundant eosinophils. Microscopy showed signs of eosinophilic pneumonia and detached allergic mucin. The patient received oral corticosteroids and aggressive bronchopulmonary toilette with resolution of symptoms.

Respiratory Institute, Cleveland Clinic Foundation, Cleveland, OH

Disclosure: The authors have no conflicts of interest to report.

Reprints: Gustavo Cumbo-Nacheli, MD, Respiratory Institute, Cleveland Clinic Foundation, Cleveland, OH (e-mail: cumbong@ccf.org).

Received October 22, 2010

Accepted December 6, 2010

Chronic eosinophilic pneumonia (CEP) is a rare disease characterized by eosinophilic infiltration of the lung parenchyma. The disease usually manifests clinically as an insidious onset of nonspecific respiratory symptoms, then progresses to include shortness of breath, bronchospasm, and cough. The bronchoscopic findings of CEP have not been well characterized. We report a case of CEP with diffuse endobronchial mucous plugging, in which bronchoscopy played an important role in the diagnosis and treatment of the disease.

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CASE REPORT

A 47-year-old woman with history of allergic sinusitis, migraines, and bronchial asthma, presented with dyspnea, cough, and yellow sputum production of 6 months duration. There had been no new exposure or identifiable trigger for the symptoms. The patient had produced thick mucous plugs in the past. Her physical examination was unremarkable, except for diffuse wheeze and pale nasal membranes. Peripheral blood showed absolute eosinophilia (3.08×103/μL, 22%). Extensive rheumatologic workup was negative. Chest computed tomography showed tree-in-bud pattern, centrilobular nodules, and bilateral peripheral pulmonary infiltrates. There was insufficient symptomatic improvement with antibiotics, inhaled albuterol, and steroids. Persistence of bilateral lung infiltrates prompted a bronchoscopic examination, which revealed extensive mucous plugging throughout the tracheobronchial tree (Figs. 1, 2). Bronchioalveolar lavage showed eosinophilia (18% of the total cell count). A cytopathologic diagnosis of eosinophilic pneumonia was made. Removal of the mucous plugs during bronchoscopy, followed by aggressive bronchopulmonary toilette and oral steroids improved her symptoms.

FIGURE 1

FIGURE 1

FIGURE 2

FIGURE 2

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DISCUSSION

CEP, also known as Carrington's disease, is a rare disorder.1 The exact prevalence remains unknown, although it has been estimated at 0.1 per 100,000 population/year.2 In adults, CEP affects women more frequently (2:1 ratio), whereas in the pediatric population boys are affected in a 1.6:1 ratio.2,3 In CEP, symptoms have an insidious onset and include wheeze, dyspnea, cough, fever, and weight loss.2,4

Suggested diagnostic criteria for idiopathic CEP include (a) nonspecific, asthma-like respiratory symptoms for more than 2 weeks; (b) peripheral pulmonary infiltrates, often bilateral; (c) alveolar and/or blood eosinophilia; and (d) exclusion of other etiologies prompting eosinophilia.5

Mucous plugs are a frequent finding in patients with cystic fibrosis, bronchiectasis, and bronchial asthma.6 Although the frequency and pathophysiology behind this phenomenon remain obscure, anecdotal reports associate mucoid plugs with eosinophilic pneumonia.7–9 It has been hypothesized that pulmonary eosinophilic infiltration prompts glandular mucous secretion; posterior water reabsorption may render bronchi obstructed by desiccated material.7 Further desquamation of bronchial epithelial lining may complicate an existing mucoid impaction.10

Our case highlights the importance of a high index of suspicion for CEP in patients with asthma-like symptoms, eosinophilia, and mucous plugging who poorly respond to bronchodilators and/or antibiotics. As in our patient, it has been described that oral corticosteroids markedly improve symptoms; treatment discontinuation usually results in symptomatic relapse.2,3

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REFERENCES

1. Carrington CB, Addington WW, Goff AM, et al. Chronic eosinophilic pneumonia N Engl J Med.. 1969;280:787–798
2. Marchand E, Cordier JF. Idiopathic chronic eosinophilic pneumonia Orphanet J Rare Dis.. 2006 6;1:11
3. Wubbel C, Fulmer D, Sherman J. Chronic eosinophilic pneumonia: a case report and national survey Chest.. 2003;123:1763–1766
4. Jederlinic PJ, Sicilian L, Gaensler EA. Chronic eosinophilic pneumonia. A report of 19 cases and a review of the literature Medicine (Baltimore).. 1988;67:154–162
5. Cottin V, Cordier JF. Eosinophilic pneumonias Allergy.. 2005;60:841–857
6. Minakuchi M, Niimi A, Matsumoto H, et al. Chronic eosinophilic pneumonia: treatment with inhaled corticosteroids Respiration.. 2003;70:362–366
7. Xie LX, Mo GX, Chen LA, et al. Chronic eosinophilic pneumonia with mucous plugs: case report Chin Med J.. 2006;119:262–264
8. Cakir E, Aksoy F, Cakir FB, et al. Chronic eosinophilic pneumonia with mucous plugs in a child Pediatr Pulmonol.. 2010;45:1040–1042
9. Warren WP. Eosinophilic reactions in the lung N Engl J Med.. 1969;281:50
10. Danila E, Norkūnienė J, Nargela R, et al. Manifestation peculiarities of idiopathic chronic eosinophilic pneumonia Cent Eur J Med.. 2010;5:75–82
Keywords:

mucoid plug; eosinophilic pneumonia; Carrington's disease

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