Bronchogenic Cyst and Its Progress in a Premature Infant

Abstract

The progressive course of a congenital bronchogenic cyst in a very low birth weight infant with respiratory distress is presented. A bronchogenic cyst, while uncommon, should be in the differential diagnosis of pneumomediastinum or medial pneumothorax even in premature infants who are on ventilators.Bronchogenic cysts are an uncommon congenital malformation of the airway with the potential for serious morbidity or mortality. There have been no previous reports of bronchogenic cysts in premature infants. Two reviews of bronchogenic cysts over a 30-year period do not mention their presence or progression.1,2 We report a case of bronchogenic cyst in a very small preterm infant and its gradual progress as shown by chest radiography. CASE REPORTWB was a 25-week gestation, 625-gm infant, born to a 26-year-old multigravida mother. Maternal history was significant for a positive tuberculin test, negative chest radiograph for infiltrates suggestive of tuberculosis, and a history of cocaine abuse. Apgar scores were 1, 5, and 6 at 1, 5, and 10 minutes, respectively. The infant was intubated in the delivery room secondary to his prematurity. He received surfactant at 1, 12, and 24 hours of life. The chest radiograph results were consistent with respiratory distress syndrome, which improved by day 3 of life ( Figure 1). On day 4 of life, a mediastinal air shadow was observed on a routine chest radiograph (Figure 2). The infant also demonstrated persistent metabolic acidosis with increasing ventilatory requirements. Based on clinical presentation, a patent ductus arteriosus was suspected. The next day, a routine radiograph of the chest showed the previously described air shadow, which was slightly increased in size.The patent ductus arteriosus was treated with three doses of indomethacin. Echocardiography obtained after treatment of the patent ductus arteriosus showed a cystic structure behind the left and right atria. A barium esophagram was performed that ruled out esophageal cyst and duplication. Despite low settings on the mechanical ventilator, the air shadow showed a progressive increase in size and a diagnosis of bronchogenic cyst was entertained (Figure 3). Cardiothoracic surgery was consulted and elective surgery was planned for the next day as the infant was stable on the ventilator with pressures of 16/5 mm Hg peak inspiratory rate (PIP/peak end-expiratory pressure), ventilatory rate of 28, and FIO 2 of 0.30. On the same night, the infant started having frequent episodes of oxygen desaturation, and a chest radiograph showed the development of a left-sided pneumothorax. A repeat radiograph obtained after inserting a chest tube showed a me-