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Extralobar Pulmonary Sequestration with Accompanying Ipsilateral Diaphragmatic Eventration

Congenital anomalies of the lower respiratory tract are rare, with pulmonary sequestrations comprising 0.15 to 6.4 per cent of all congenital pulmonary malformations.1 Pulmonary sequestrations can be categorized as either extralobar or intralobar sequestrations. The former are distinct from the normal lung tissue with a separate pleural lining and represent 15 to 25 per cent of all pulmonary sequestrations. The later are found within normal lung tissue. Each has an anomalous systemic blood supply and no bronchial communication with the normal tracheobronchial tree. Extralobar sequestrations are more frequently found to have associated anomalies as compared with intralobar sequestrations.2 This case represents an asymptomatic patient with prenatal diagnosis of left pulmonary sequestration found to have a simultaneous ipsilateral diaphragmatic eventration. A 12-month-old male presented to the pediatric surgery service with prenatally diagnosed left pulmonary sequestration. Subsequent postnatal chest CT demonstrated cystic change in the left lower lobe with both mass effect and aberrant vasculature raising concern for a hybrid lesion including sequestration and congenital pulmonary adenomatoid malformation. An additional finding of a possible congenital diaphragmatic hernia was noted given an abnormally superior position of the left kidney. The patient underwent left thoracoscopy with conversion to left thoracotomy due to intraoperative findings of an extrapulmonary sequestration with large ectopic vasculature as well as a large posterolateral diaphragmatic eventration in the position of a Bochdalek hernia (Fig. 1). There was continuity of the diaphragm without herniation, and this was the obvious source of the elevated left hemidiaphragm and ectopic left kidney. The extralobar pulmonary sequestration was initially mobilized allowing for identification of dual arterial supply emanating from below the diaphragm and an aberrant venous drainage (Fig. 2). The left lower lobe remained well aerated and because of the very small cystic changes in the lung on CT scanning, we elected to forgo a left lower lobectomy. The large eventration was then plicated to allow for complete repair of the left hemidiaphragm. Bronchopulmonary foregut malformations encompass pulmonary lesions such as congenital cystic adenomatoid malformation, congenital lobar emphysema, sequestration, bronchogenic cyst, and tracheal/bronchial atresia.3 These malformations are hypothesized to result from the disturbance in the growth and development of the pulmonary system through abnormal budding of the ventral region of the embryonic foregut that develops into the tracheobronchial tree. Pulmonary sequestrations represent a small subsection of these lesions and can be divided into intralobar and extralobar forms. The former are continuous with the surrounding lung parenchyma with shared pleura, whereas the latter are separate from the normal lung with a separate pleural lining.1 Extralobar sequestrations are often asymptomatic, as in this patient, and may be discovered during another operative procedure.