Pulmonary Eosinophilic Granuloma Complicated by Recurrent,Bilateral Pneumothoraces: A Case Report

Abstract

Pulmonary eosinophilic granuloma (EG) is a rare disease of unknown etiology typically presenting with mild symptoms of cough or dyspnea, which usually resolve spontaneously. However, occasional cases have a more fulminant course resulting in extensive lung destruction and respiratory failure. Chest radiographs characteristically demonstrate reticulonodular or cystic changes, and pneumothoraces complicate pulmonary EG in 10% to 20% of cases. No therapy has been shown to be effective. We report a previously healthy 19-year-old male who developed rapidly progressive respiratory failure and bilateral pneumothoraces due to pulmonary EG. Persistent alveolar leaks and recurrent pneumothoraces posed major management problems despite an aggressive surgical approach. During a 7-week hospitalization period, the patient required 21 thoracostomy tubes and underwent thoracotomy and surgical pleurodesis three times. The patient was discharged with full expansion of the left lung and a small right pneumothorax. In the months following discharge, despite two brief periods of rehospitalization due to enlargement of the right pneumothorax, his dyspnea lessened and his exercise tolerance gradually improved. One year after the onset of symptoms, he reported continued improvement and a chest radiograph showed fewer infiltrates and no evidence of pneumothorax or residual bullae. We suggest that an aggressive surgical approach is required for patients with pneumothorax complicating EG. We recommend a standard posterolateral thoracotomy to expose the entire pleural surface, the stapling and excision of all blebs and bullae, and the vigorous abrasion of the entire pleural surface. (Respir Care 1985;30:1049-1056.)