Symptomatic bronchopulmonary disorders have been only occasionally reported in Crohn's disease, although several studies have documented the possibility of latent involvement of the respiratory tract. We report the case of a patient with long-standing Crohn's disease who presented with acute transient chest pain and a recent history of mild dyspnea and nonproductive cough. Chest radiographs were normal, while high-resolution computed tomography demonstrated a mosaic pattern of attenuation that was consistent with a bronchiolar disorder. Pulmonary function tests showed mild airway obstruction and normal diffusion indices. Thoracoscopic lung biopsy demonstrated focal infiltration of the bronchiolar walls by mononuclear cells and non-necrotizing granulomas. To our knowledge, this is the first account of isolated granulomatous bronchiolitis in Crohn's disease. These findings suggest that a granulomatous inflammatory process of the bronchioles could be involved in the development of airway obstruction in patients with Crohn's disease.