Congenital abnormalities of the large airways are uncommon, but may occasionally pose significant difficulties for anesthesiologists. The tracheal bronchus is an anatomical variant in which an accessory bronchus originates directly from the trachea rather than distal to the carina, as a takeoff from the right mainstem bronchus. Anesthesiologists should be aware of this uncommon anomaly, its different variants, and its management in order to successfully establish one lung ventilation (OLV) for surgical isolation. In this article, we report the challenges encountered in establishing OLV in a patient with a previously undiagnosed aberrant right upper lobe bronchus arising directly from the trachea.