Endobronchial leiomyomas are rare benign tumors, accounting for less than 2% of all benign lung tumors. Known to arise from the smooth muscle of the tracheobronchial tree, leiomyomas occur in parenchymal, endotracheal, or endobronchial locations. Endobronchial lesions constitute about 33% of all pulmonary leiomyomas. Literature on primary endobronchial leiomyomas is extremely limited. Symptomatology depends on the degree of endoluminal bronchial obstruction.We report a case of 71-year-old woman admitted to our hospital complaining of persistent non-productive cough. Computed tomography (CT) imaging revealed a rounded tumor arising in the right upper lobe bronchus which subsequently underwent bronchoscopy and tissue sampling for definitive treatment. We also describe the cytological characteristics of endobronchial pulmonary leiomyoma.