Atelectasis With Peripheral Eosinophilia, in a Patient With Metastatic Melanoma

Abstract

INTRODUCTION: INTRODUCTION: Bronchocentric granulomatosis is an injury pattern characterized by granuloma formation and necrosis centered around and limited to the bronchi and bronchioles. The disease entity is defined by morphologic criteria and is not a clearly defined clinical syndrome. CASE PRESENTATION: A 72 - year -old female former smoker with a history of melanoma, presented to our institution in January 2012 with progressive dyspnea on exertion, following a sinus infection in December. Her dyspnea progressed despite a course of azithromycin and further treatment with fluticasone/salmeterol and albuterol at her local emergency room. Her past medical and surgical history includes melanoma of the right upper extremity (1993) with metastasis to the axilla and supraclavicular region, treated with excision, high dose interferon, and radiation. Video-assisted thoracoscopic right lower lobectomy, in February 2011 indicated for a suspicious pulmonary nodule, showed necrotizing and non-necrotizing granulomatous inflammation. She is a former smoker (2 pack years) without known tuberculosis, animal or significant occupational exposures. Her most recent travel was to Honduras in 2009. She reported significant food allergies and an episode of bronchospasm and erythema, following a prior dose of penicillin. Physical examination was unremarkable. Chest x-ray on admission revealed collapse of the right upper lobe (Figure 1) confirmed by CT scan of the chest, which demonstrated impaction and opacification of the right upper lobe bronchus and rightward mediastinal shift. Lab data: WBC 5.9, eosinophils 26.8, IgE level 960. Fungal serologies, aspergillus antibodies, and connective tissue workup was negative. Angiotensin converting enzyme levels and pulmonary function tests were within normal limits. Bronchoscopy revealed necrotic material extending from the right upper lobe. Pathology demonstrated allergic layering of mucin and eosinophilic cellular debris containing Charcot -Leyden crystals (Figure 2). The patient was started on daily prednisone and sulfamethoxazole/trimethoprim prophylaxis, with marked improvement in symptoms and resolution of right upper lobe collapse on a follow up chest x-ray, one month later. DISCUSSION: Bronchocentric granulomatosis has various etiologies including: allergic bronchopulmonary aspergillosis, fungal infections and connective tissue disorders. Steroids are indicated if the patient is acutely hypoxemic or in distress. Patients often undergo a lobectomy for suspicion of malignancy, but there is no clear benefit to surgical excision. CONCLUSION(S): This is a unique case of steroid responsive bronchocentric granulomatosis of unclear etiology, resulting in marked improvement of central airway obstruction and lobar collapse after treatment.