Thoracoscopic resection is being used more commonly for the treatment of congenital cystic adenomatoid malformation in neonates and infants. However, in the rare case of congenital cystic adenomatoid malformation with congenital absence of lung fissure, thoracoscopic lobectomy cannot be performed safely. Moreover, if the lung lesion is deep-seated and cannot be visualized on the pleural surface, wedge resection may result in residual lesion. Here, we reported our approach of thoracoscopic resection under hookwire guidance to tackle this problem.