Gallbladder perforation is an extremely rare condition in infants. We present a premature infant who had congestive heart failure and renal failure owing to complex congenital heart disease and developed gallbladder perforation at 60 days of age. The patient showed neither signs of peritonitis nor impaired liver function; however, we found bile drainage from the peritoneal dialysis tube. Emergent laparotomy revealed a 3-mm perforation with surrounding tissue necrosis over the gallbladder neck. Cholecystectomy was performed after patent common bile duct was demonstrated using intraoperative cholangiography. No stones were noted in the gallbladder. Further histologic examinations of the perforation site showed ischemic changes. There were no biliary complications after surgery. Nevertheless, the patient died of multiple organ failure at 120 days old.