Pulmonary Langerhans cell histiocytosis (LCH) is a rare disease. The typical findings on chest computed tomography (CT) of this disease are multiple bizarre cystic spaces associated with small peribronchiolar nodules, predominantly in the upper third of both lungs. The peribronchiolar nodules are not always present due to the evolution sequence. For patients with histiologically proven extrapulmonary LCH, the diagnosis of pulmonary LCH is usually established if the findings on high-resolution CT are consistent with pulmonary LCH. Recently we encountered one patient with histiologically proven osseous LCH. The chest CT feature of this patient was multiple bizarre cystic spaces without obvious pulmonary parenchymal nodule.