Objective: Congenital cystic adenomatoid malformation (CCAM) in adults is very rare, and surgical resection is the treatment of choice. We evaluated the clinical characteristics and feasibility and safety of video-assisted thoracic surgery (VATS) in adult patients with CCAM.
Methods: Retrospective analysis of nine consecutive adult patients with CCAM treated surgically between January 1995 and April 2006 at a single center. The clinical characteristics and surgical outcome were evaluated retrospectively.
Results: The records of five men and four women were examined. The median age at diagnosis was 30.5 years (range 16-44 years). The most frequent symptoms were cough (n=4) and hemoptysis (n=2). Three patients had recurrent respiratory infections. Chest computed tomography (CT) was reviewed in all cases. The lesions were on the right side in six patients and on the left in three. All patients had lesions in a single lobe, except one patient who had multi-lobar lesions. The CCAM appeared as thin-walled multi-septated cystic lesions in six patients and as parenchymal consolidations in three patients. Four patients underwent VATS lobectomy and four underwent lobectomy through a conventional open thoracotomy. One patient had open pneumonectomy. The median operating time (160 min in VATS lobectomy and 165 min in open lobectomy), median duration of chest tube drainage (4 days in VATS lobectomy and 6.5 days in open lobectomy), and postoperative complications (one patient in each treatment group) were similar between the VATS and open lobectomy groups. The hospital stay was shorter in the VATS lobectomy group (median 5 days) than in the open lobectomy group (median 7.5 days). No postoperative or procedure-related mortality occurred.
Conclusions: CCAM is rare in adults. Patients usually present with recurrent respiratory infections and thin-walled multi-septated cystic lesions. VATS is a feasible and safe technique to treat adult patients with CCAM.