BACKGROUND Bronchiolitis obliterans is the term used to describe a clinical syndrome of irreversible airflow obstruction. Among the etiologies linked to this entity is the rarely reported association with Stevens-Johnson syndrome, which has had a poor outcome in most of the previously published cases. The optimum management of bronchiolitis obliterans as a complication of Stevens-Johnson syndrome is not well defined. CASE REPORT A 41-year-old woman developed significant shortness of breath 3 months after recovering from Stevens-Johnson syndrome precipitated by a second-generation cephalosporin. She was found to have severe irreversible airway obstruction on physiology studies, and computed tomography scans of the inspiratory and expiratory phases of respiration showed air trapping that was more prominent on expiratory films. The patient was diagnosed with bronchiolitis obliterans, for which bronchodilators and long-term macrolide therapy were administered. Although she did not recover completely, her follow-up physiology studies showed that the bronchiolitis obliterans was stable. CONCLUSIONS Bronchiolitis obliterans secondary to Stevens-Johnson syndrome is a rare entity that is progressive and can lead to functional impairment. Identifying the disease at an early stage might stabilize or slow its progression. Herein, we describe a case of bronchiolitis obliterans as a complication of Stevens-Johnson syndrome and review the literature to raise awareness of this condition, highlight its course, and discuss the available treatments.