Acquired subglottic cyst in infancy is almost always associated with episodes of early life intubation. Most cases typically presented late, usually days to months after extubation. We report a case of a subglottic cyst with different presentation than the norm. This case highlights that subglottic cyst can present acutely, and rapidly enlarging soon after the airway extubation. As the management of a large subglottic cyst can be challenging, a close observation for early diagnosis and intervention are recommended post extubation in the high-risk cases, such as in the premature infant.