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Tracheo-innominate artery fistula (TIAF) is rare, yet the most fatal complication after tracheostomy. In the absence of immediate diagnosis and surgical management, the mortality rate is very high, because the complication can lead to sudden massive tracheal hemorrhage. Tracheal obstruction and hypovolemic shock are the major life threatening conditions. The 46-year-old woman received tracheostomy tube insertion after stroke. Three months later, there was occurrence of active bleeding at the site of tracheostomy in the patient, who participated in comprehensive rehabilitation program. Immediately, the patient received an endotracheal tube insertion into the tracheostomy site and thus massive bleeding was controlled. The patient was transferred to the intensive care unit, where her breathing was maintained by mechanical ventilation. Based on computed tomography and laryngoscopy, no remarkable findings about TIAF were detected. Nevertheless, transfemoral angiography findings revealed that innominate artery made small luminal outpouching to trachea at the carotid artery and at the subclavian artery bifurcation level, based on which a diagnosis of TIAF was made. She had an operation for TIAF, tracheoplasty with bypass graft. Subsequently, she was discharged after 15 weeks. In the present report, we describe a case of TIAF, which can occur in the patients with tracheostomy tube during rehabilitation.