Background. Single-incision laparoscopic surgery has been increasingly adopted in pediatric surgery. Nevertheless, its feasibility and safety in neonates with congenital biliary malformations is unclear. This study reports successful single-incision laparoscopic hepaticojejunostomy (SILH) for neonates with extrahepatic biliary cystic lesions. Methods. Ten neonates with extrahepatic biliary cystic lesions (choledochal cyst/correctable biliary atresia: 6/4) who underwent SILH between May 2011 and September 2011 were reviewed. Ultrasonography, upper gastrointestinal contrast studies, and laboratory tests were performed during the follow-up period. Results. Mean operative time, postoperative hospital stay, time to full feeding, and duration of drainage were comparable to our historic open-control groups of 15 neonates with choledochal cysts and 7 patients with correctable biliary atresia. Median follow-up duration was 6.0 months. They regularly took medical treatments during the follow-up periods. The jaundice subsided or liver function was normalized within 3 months postoperatively. So far, no mortality or morbidity of cholangitis, bile leak, anastomotic stenosis, and intrahepatic reflux were encountered. Conclusions. In experienced hands, SILH for neonates with extrahepatic biliary cystic lesions is feasible and safe. It provides a new alternative for neonatal hepatobiliary surgery.