Endobronchial leiomyoma is extremely rare. Most endobronchial leiomyomas reported in the literature have been resected by either lobectomy or pneumonectomy. We herein report a case treated by sleeve bronchoplasty without pulmonary resection. A 42-year-old woman was admitted to our hospital complaining of hemoptysis. Bronchoscopy revealed a lobulated tumor arising from the medial wall of the right main stem bronchus. A sleeve resection of the right main bronchus including the tumor and end-to-end anastomosis was performed. The histological diagnosis of the resected specimen was leiomyoma with no evidence of malignancy. The importance of early diagnosis and appropriate surgical treatment to preserve pulmonary function are emphasized. Similar cases of an endobronchial type of pulmonary leiomyoma reported in the literature are also reviewed.