Background Several reports have documented that the pulmonary sequestration is in communication with the gastrointestinal tract and the concept of bronchopulmonary foregut malformation (BPFM) has become more widespread. However, there are few reports of the sequestration associated with the pancreas derived from the foregut. We describe the history and pathophysiology of BPFM including pancreatic tissue in a male infant with respiratory distress. Case presentation A male patient was born at 38 weeks of gestation and weighed 2752 g at birth. He developed pneumonia and was hospitalized at 3 months of age. Chest radiographs and CT scans led to the diagnosis of a lung abscess in the left lower intralobar pulmonary sequestration with aberrant arteries from the abdominal cavity. At 4 months of age, when the abscess had resolved, left lower lobectomy and the resection of the intralobar sequestration were performed. The pulmonary sequestration was conjoined with the esophagus. A fistula was found between the lower esophageal wall and the pulmonary sequestration. An additional small segment of the esophageal wall was excised. Histologically, the mediastinal surface of the sequestration tissue contained pancreatic tissue. Furthermore, esophageal and gastric tissue, cartilage tissue, and ciliated epithelium were confirmed. A definitive diagnosis of BPFM was made. Conclusions We postulated the rare case of a communicating BPFM with intrapulmonary sequestration on one end and the esophagus on the other forming a mass lesion, which included ectopic pancreatic tissue in a male infant.

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Background: Acquired cystic lung disease is a serious respiratory complication of bronchopulmonary dysplasia in premature infants. Most cases of acquired cystic lung disease underlying bronchopulmonary dysplasia involve pulmonary interstitial emphysema. Although this is a reversible condition, there are a few instances where surgery might be necessary. An accurate diagnosis is important to decide the therapeutic strategy for acquired cystic lung disease. Here, we report a rare case of a giant pulmonary bulla in an infant treated with bullectomy. Case presentation: A male infant born at 23 weeks of gestation with a birth weight of 524 g was initially diagnosed with respiratory distress syndrome. During mechanical ventilatory support, he presented with recurrent pneumothorax and a gradually expanding pulmonary cyst in the right lung. Chest CT at 5 months of age revealed a large cyst located in the subpleural area adjacent to the multiple cystic air spaces. These findings are consistent with the diagnosis of giant pulmonary bulla with pulmonary interstitial emphysema underlying bronchopulmonary dysplasia . At 9 months of age, the giant pulmonary bulla expanded further due to acute bronchitis for which he developed respiratory failure and obstructive shock. This warranted a bullectomy for the giant pulmonary bulla. After the operation, the unresected pulmonary interstitial emphysema lesion did not expand further. He is currently three years old and has no respiratory problems. Conclusions: This case demonstrated that chest CT is useful for providing valuable anatomical information necessary in deciding the treatment strategy for acquired cystic lung disease in infants.

Acquired cystic lung disease in premature infants is a serious respiratory complication, and pulmonary interstitial emphysema (PIE) has been widely reported. We report a rare case of giant pulmonary bulla in an infant treated with bullectomy where chest computed tomography was useful in directing treatment.