We present a case of symptomatic complex bronchopulmonary foregut malformation (BPFM), including extralobar pulmonary sequestration and a bronchogenic cyst, in the left anterior mediastinum of a 15-year-old boy. Preoperative computed tomography showed a cystic mass with heterogeneous enhancement of adjacent soft tissue components and pleural effusion. We suggested the infected bronchogenic cyst as the first impression. However, pathological examination after surgical resection revealed extralobar pulmonary sequestration and a bronchogenic cyst with unusual manifestation, which was located in the left upper hemithorax and supplied by the pulmonary artery. In patients presenting with a cystic mass with features of inflammation or infection and collateral vasculature, the possibility of a complex bronchopulmonary foregut malformation should be considered in the differential diagnosis. J Thorac Dis 2017;9(7):E632-E635 jtd.amegroups.com suggesting an extrapulmonary origin. The mass contained a non-enhanced homogenous low-attenuated fluid component ( Figure 1A). There was a scanty amount of pleural fluid in the left hemithorax, suggesting perforation of the cystic mass ( Figure 1A). A small artery originating from the left pulmonary artery and entering the medial side of the cystic mass was visualized ( Figure 1B). We therefore suggested the presence of a complicated cystic mass, such as a bronchogenic cyst or dermoid cyst, with infection.The patient underwent thoracoscopic surgical intervention for lesion resection. During surgery, we found a supernumerary lobe of the left lung, with a vascular hilum, between the upper and lower lobes. This lobe consisted of cystic and solid components with hilar vasculature without perforation. The cystic component, which contained thick mucus-like material, included the hilum, and the solid component, which turned out to be composed of lung tissue, was an inclusion of the cystic mass ( Figure 1C). A small feeding vessel originating from the hilum was identified and divided with an endoscopic stapler. Gross pathological examination showed a unilocular cystic mass within the inflamed sequestrated lung ( Figure 1D) with a wall showing fibrotic change. Microscopically, a bronchial
DiscussionBPFMs have been described by numerous classifications and terminologies incorporating a common origin and association. These include congenital anomalies associated with the development of the foregut (bronchogenic cyst, esophageal/neurenteric cyst, tracheoesophageal fistula/ diverticula/stenosis), pulmonary [pulmonary agenesis/ hypoplasia, congenital lobar emphysema, congenital pulmonary airway malformation (CPAM)], airway (tracheal/ bronchial atresia, tracheal bronchus), and vascular (pulmonary sling, alveolar capillary dysplasia) components (1,4,5).Bronchogenic cysts generally demonstrate an enhanced capsular wall and a homogenous consistency on enhanced CT because they have a bronchial epithelial lining and are filled with mucus (6). In the case of complicated bronchogenic cysts, a th...