Leiomyoma of trachea is one of the rarely encountered benign tumors, accounting for about 4% of all benign tracheal tumors. A 69-year-old woman with Diffuse Large B-cell lymphoma (DLBCL) was found on PET to show tracer accumulation in the trachea. She had no symptoms, and we identified the tumor in her trachea by fiberoptic laryngoscopic examination. We performed tracheotomy and tumorectomy under local anesthesia. Histopathological examination of the resected tumor revealed spindle-shaped tumor cells with mild nuclear atypia, proliferating in an interlacing pattern in the subepithelial layer, and a few mitotic figures in the nuclei; immunostaining revealed positive staining results for α-SMA and desmin. Until now, 8 months since the surgery, she has shown no signs of tumor recurrence. We had exenterated the tracheal leiomyoma safely and she is now living leiomyoma-free.