Aspergillus tracheobronchitis (ATB) represents a rare disease entity accounting for 3.5% to 5.0% of cases of invasive aspergillosis. Particularly, mass-forming ATB is extremely rare, and clinical reports are limited. Given that the patency of the trachea and bronchi are essential for maintaining sufficient airflow, a central airway mass originating from an Aspergillus infection is likely to be associated with severe clinical manifestations and fatal outcomes. Although several therapeutic options for ATB have been suggested, including medication and airway interventions, the optimal choice of treatment in diverse clinical conditions remains under discussion.We report a case of ATB that initially manifested as severe dyspnea and total atelectasis of the left lung in a patient with newly detected diabetes.Radiographic study, bronchoscopy, and pathologic findings of the lesion revealed mass-forming type of ATB.Interestingly, our patient's symptoms dramatically resolved with voriconazole without further invasive intervention.This clinical experience highlights the beneficial role of voriconazole in the treatment of rare and potentially fatal cases of ATB.