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Chronic Eosinophilic Pneumonia With Endobronchial Involvement

Hara, Johsuke MD, PhD*; Nishi, Kouichi MD, PhD*; Demura, Yoshiki MD, PhD*; Kurokawa, Kohji MD*; Kurumaya, Hiroshi MD, PhD; Katayanagi, Kazuyoshi MD, PhD; Kasahara, Kazuo MD, PhD; Fujimura, Masaki MD, PhD; Nakao, Shinji MD, PhD

Journal of Bronchology & Interventional Pulmonology: July 2011 - Volume 18 - Issue 3 - p 285–287
doi: 10.1097/LBR.0b013e318222a05d
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We report here a case of chronic eosinophilic pneumonia with significant endobronchial involvement. A 59-year-old man was admitted complaining of fever, productive cough, wheezing, and dyspnea. There were ground-glass opacities in bilateral upper fields and tram track shadows in the left lower lung field on chest x-ray, and ground-glass opacities in bilateral upper lobes, thickening of the bronchial walls, and centrilobular nodules on computed tomographic scan of the chest. There was marked eosinophilia in the peripheral blood and bronchoalveolar lavage fluid and was diagnosed as chronic eosinophilic pneumonia. Bronchoscopy also revealed white nodules at the orifice of the right S6 and the left S1+2 bronchi. The histologic examination of these nodules revealed eosinophilic inflammation into the bronchial wall. This is a rare case of chronic eosinophilic pneumonia with endobronchial eosinophilic involvement.

*Department of Respiratory Medicine

Department of Pathology, Ishikawa Prefectural Central Hospital

Department of Respiratory Medicine, Cellular Transplantation Biology, Kanazawa University Graduate School of Medicine, Kanazawa, Ishikawa, Japan

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

Reprints: Johsuke Hara, MD, PhD, Department of Respiratory Medicine, Ishikawa Prefectural Central Hospital, 2–1, Kuratsuki-Higashi, Kanazawa, Ishikawa, 920-8530, Japan (e-mail: hara-jam@violin.ocn.ne.jp).

Received February 12, 2011

Accepted February 21, 2011

Eosinophilic lung diseases are a heterogeneous group of disorders. Both acute eosinophilic pneumonia (AEP) and chronic eosinophilic pneumonia (CEP) are known to be complicated with eosinophilic inflammation in central airway.1,2 In contrast, there had been few reports regarding bronchoscopic findings of airway lesions of eosinophilic pneumonia. We present a rare case of eosinophilic pneumonia with endobronchial eosinophilic lesions.

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CASE REPORT

A 59-year-old man was admitted during December 2007 complaining of fever, productive cough, wheezing, and dyspnea. He had been diagnosed with bronchial asthma at another institution 1 year earlier. Physical examination revealed a temperature of 37.9°C and inspiratory and expiratory wheezes in all lung fields. There were ground-glass opacities in bilateral upper fields and tram track shadows in the left lower lung field on chest x-ray (Fig. 1), and ground-glass opacities in bilateral upper lobes, thickening of the bronchial walls, and centrilobular nodules on chest computed tomographic scan of the chest (Fig. 2). Laboratory tests showed marked eosinophilia (white blood cells 15,510/μL with 37.9% eosinophils). C-reactive protein was 5.0 mg/dL, erythrocyte sedimentation rate was 74 mm/h, and total IgE level was 859.4 IU/mL. Radioallergosorbent testing showed that specific IgE antibodies to house dust, Dermatophagoides pteronyssinus, Dermatophagoides farinae, Aspergillus, Candida, Alternaria, cedar pollen, ragweed, orchard grass, and sweet vernal grass were negative. Myeloperoxidase-antineutrophil cytoplasmic antibody was negative. Arterial blood gas analysis during breathing room air revealed PaO2 60.1 Torr, PaCO2 38.6 Torr, and A-aDO2 (alveolar-arterial O2 difference) 36.2 Torr. Differential cell analysis of sputum revealed 80% eosinophils. Cultures for microorganisms from the patient's sputum grew normal flora. A pulmonary function test, according to the standards of the American Thoracic Society,3 revealed FVC of 2.40 L (64.3% of predicted), FEV1 of 1.28 L (40.5% of predicted), an FEV1/FVC ratio of 53.3%, TLC of 5.91 L (101.9% of predicted), RV of 2.97 L (164.1% of predicted), and RV/TLC ratio of 50.27%, DLCO of 19.47 mL/min/mm Hg (91.6% of predicted), and gas transfer coefficient (DLCO/VA) of 4.33 mL/min/mm Hg/L (91.5% of predicted). Differential cell analysis of bronchoalveolar lavage fluid recovered from the right B3 revealed 99.0% eosinophils, 1.0% lymphocytes, and an increased total cell count of 47.5×105 cells/mL. On the basis of these results, we diagnosed the patient with CEP. Furthermore, bronchoscopy revealed white nodules at the orifice of the right S6 segment (Fig. 3). The same nodular lesions were also found at the orifice of the left S1+2 Segments. The histologic examination of those nodular lesions revealed eosinophilic inflammation into the bronchial wall with thickening of basal membrane (Fig. 4).

FIGURE 1

FIGURE 1

FIGURE 2

FIGURE 2

FIGURE 3

FIGURE 3

FIGURE 4

FIGURE 4

Prednisolone (30 mg/d) was prescribed, resulting in a rapid symptomatic and radiographic improvement. The white nodules documented by the bronchoscopy also disappeared after 7 days of prednisolone treatment. Pulmonary function test revealed FVC of 5.48 L (146.8% of predicted) and FEV1 of 3.52 L (111.4% of predicted) after 14 days of prednisolone treatment. Improvement and relapse of his symptoms occurred synchronously during the course of tapering of prednisolone. Although obstructive ventilatory impairment remained despite the therapy, he was doing well subjectively on maintenance treatment with prednisolone (10 mg/d).

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DISCUSSION

Eosinophilic lung diseases are classified into simple pulmonary eosinophilia (Löffler syndrome), AEP, CEP, idiopathic hypereosinophilic syndrome, asthma, allergic bronchopulmonary aspergillosis, bronchocentric glanulomatosis, Churg-Strauss syndrome, parasitic infections, and drug reactions.4 CEP was individualized as a distinct entity by Carrington in 1969.5 Several researchers have reported that the patients with CEP developed bronchial asthma before or after the diagnosis of CEP.6–9 Marked infiltration of eosinophils into the bronchial mucosa and submucosa, which was observed in the specimens obtained from central airways by bronchial biopsy was also reported in 2 patients with CEP.2 Furthermore, in the patients with AEP, specimens of bronchial wall revealed eosinophil infiltration into the central airway.1 In contrast, to our knowledge, endobronchial eosinophilic lesions associated with eosinophilic lung disease are very rare; so far, 3 patients with bronchial asthma,10–12 2 patients with Churg-Strauss syndrome,13 and 1 patient with hypereosinophilic syndrome14 are reported in the literature. In these reports, endobronchial lesions had marked infiltration of eosinophils. Endobronchial lesions disappeared with systemic corticosteroids,14 systemic corticosteroids and cyclophosphamide,13 or inhaled corticosteroids.12 In our case, the nodular lesions showed inflammatory cell infiltration mainly consisting of eosinophils into the bronchial mucosa and submucosa and resolved with systemic prednisolone. The cause of endobronchial lesions remains unclear. Matsushima et al13 speculated that severe allergic inflammatory reactions resulted in mucosal lesions revealing necrotizing bronchial inflammation with many eosinophils.

This is a rare report of CEP with significant endobronchial involvement and lesions mainly consisting of eosinophils. The mechanism of bronchial lesions in CEP is unknown and reports of more cases are needed for further description.

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REFERENCES

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Keywords:

chronic eosinophilic pneumonia; eosinophilic lung disease; endobronchial involvement

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