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Purpose: Noninvasive positive pressure ventilation (NIPPV) is one of the ventilation-supporting methods by providing adequate exogenous pressure without intubation or tracheostomy. We aimed to assess the frequency and clinical factors for pneumothorax occurring during NIPPV application in a tertiary children’s hospital. Methods: We selected cases of pneumothorax related to NIPPV by keyword searching in our institution's clinical data warehouse, and their medical records were retrospectively reviewed. Results: During a period of 17 years, 15 cases undergoing NIPPV developed pneumothorax, which was an incidence of 0.64% (15 of 2,343). There were 9 neonates and 6 adolescents. In 9 neonates, pneumothorax was caused by the continuous positive airway pressure (CPAP) ventilator, and occurred on 2 days after birth (median, range 1–3 days). In neonates, pneumothorax developed within 36 hours after CPAP application. One neonate underwent tracheal intubation and 3 neonates underwent chest tube insertion. In the postteenager group, pneumothorax developed 23 months (median, range 5 days to 47 months) after NIPPV application with a mask. All of the 6 patients had underlying neuromuscular disorders and one had superimposed interstitial lung disease. One of the 7 cases underwent surgical treatment and 4 cases were intubated. One case died from the deterioration of underlying interstitial lung disease. Conclusion: Although it rarely happens, the NIPPV can result in pneumothorax. In most cases, it can be resolved by supportive cares with oxygen or chest tube with or without tracheostomy. The prognosis is related to the type of underlying disease and its progression.