After two transbronchial resections, the patient's symptoms of paroxysmal cough, whistle sound and shortness of breath were significantly reduced, as well as the airway stenosis was significantly improved. Although the features examined by enhanced chest computed tomography, positron emission tomography/computed tomography, diffusion-weighted imaging of enhanced chest MRI and bronchoscopy indicate to be malignant tumor, the patient was definitely confirmed by multiple transbronchial biopsies and immunohistochemistry. Here we present a rare case of 18-year-old male diagnosed with primary diffuse RDD in central airway. It is difficult to differentiate it from primary airway malignant tumor and to diagnose correctively in time. Central airway RDD is similar to malignant tumor in imaging by radiological method and in bronchoscopy features. It is even rarer occurring only in central airway of lung and in diffuse form. Rosai-Dorfman disease (RDD) is a rare benign non-langerhans cell histiocytosis, mainly involving lymph nodes and skin.
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