Castleman's disease (CD) in trachea is rarely reported to date. This report introduces a case of CD in trachea with emphasis on its radiological presentation onCT. A female patient was admitted at our emergency department with dyspnoea. Plain and enhanced CT revealed a mass with distinct border and marked enhancement. Tracheotomy was planned and it turned out to be CD of hyaline type on pathological examination. Two similar case reports on intratracheal CD from 1954 to 2015 were reviewed as we searched in PubMed using key words "endotracheal Castleman's disease" or "Castleman's disease in trachea" or "Castleman's disease in tracheal". This will be the third case report of CD in trachea in English literature. In this case report, the radiological appearance of CD on multiple imaging modes is reviewed. Lesions that should be taken into consideration in differential diagnosis are mentioned. The two main surgical methods for such lesions are briefly described.