Coexistence of Intrapulmonary Bronchogenic Cyst and Congenital Cystic Adenomatoid Malformation

Development of the respiratory system begins at 3 weeks gestation, and aberrations in the developmental processes may give rise to a group of structural abnormalities collectively referred to as bronchopulmonary foregut malformations (BPFMs). The common BPFMs include congenital cystic adenomatoid malformation (CCAM), pulmonary sequestration, congenital lobar emphysema, and bronchogenic cysts (BCs) and all these often present on imaging studies as abnormal cystic structures. Various types of BPFMs may occur in conjunction with one another or in association with other congenital anomalies. The coexistence of CCAM and extralobar pulmonary sequestration (ELS) has been reported, but other combined BPFMs are extremely rare. To the best of our knowledge, this is the first case report of intrapulmonary BC associated with CCAM to appear in the English medical literature with the exception of a case of coexisting ELS, CCAM and BC in a fetus. We report here on a unique case of coexisting intrapulmonary BC and CCAM in a 13-year-old female and we review the relevant medical literature.