Discussion and conclusion
CBD stone is a rare condition in children, although it has become more common recently (8). Unlike adults, it is mostly symptomatic in children depending on several factors such as age, ethnicity, geographical localization, medical facilities, and referral status (9). The most common cause of cholelithiasis in children is a hemolytic disease (20–30%). Other pathologic situations such as congenital hepatobiliary diseases, obesity, receiving total parenteral nutrition, ileal disease or resection, use of ceftriaxone, metabolic syndrome, choledochal cyst, PFIC, NEC, biliary cirrhosis, prematurity, Wilson disease, cystic fibrosis, congenital heart diseases, and idiopathic cholelithiasis should also be considered as the causes of cholelithiasis in pediatrics. (10). Houman et al. reported a 12-year-old boy with a hemolytic uremic syndrome, established by renal biopsy, who developed cholestatic jaundice. It was discovered by ERCP and extracted by sphincterotomy (11). It has been proven that for treating pancreaticobiliary disease, endoscopic sphincterotomy or CBD explorations is a safe method, even in pediatric population (12, 13). The case presented in this report was symptomatic. The patient had no history or family history of any medical conditions except prematurity. One of the important findings in the MRI and MRCP was the left-sided gallbladder. Despite stone removal, he is still at risk for CBD stone formation because of having an anomaly in the biliary system. So, the patient was advised to seek cholecystectomy in a soon future.
The left-sided gall bladder refers to a gallbladder lying on the left side of the falciform ligament (14). The reported incidence of this anomaly is estimated to be between 0.1% and 1.2% (13). It is very rare and includes three anatomic abnormalities: a right-sided ligamentum teres, an ectopic left-sided gallbladder, and a situs inversus (9). The possible associated abnormalities with the left-sided gallbladder are portal vein anomalies, biliary system anomalies, and left lobe hypoplasia (15). It has been reported that the likelihood of intraoperative bile duct injuries in individuals with left-sided gall bladder is higher than the average population (up to 7.3%) due to anomalies of the bile duct, portal vein, and other anatomical structures in the hepatobiliary system (16). The patient discussed in our report has trifurcated main portal vein with a hypoplastic ascending portion (umbilical portion) of the left portal vein. Moreover, small teres ligament differentiation of the fourth segment was suboptimal.
As mentioned before, a left-sided gall bladder is a rare condition. Nevertheless, it is possible to accurately diagnose a left-sided gall bladder before surgery and perform laparoscopic cholecystectomy by adjusting the port position. Increased size of the left portal vein and distribution of the left portal vein crossing over to the right side of the liver is the crucial common features of the left-sided gallbladder. These variations probably have considerable clinical implications in managing hepatic resection, including donor hepatectomy (17).
In recent years, the treatment approaches for managing choledocholithiasis in children have become more specific; however, no gold standard procedure is available yet. The endoscopic approach for managing biliary tract obstruction in medical centers performing ERCP is usually the first choice. Similarly, laparoscopic evaluation of CBD has also proven safe and effective. In the absence of ERCP, laparoscopic investigation can be an appropriate alternative. Laparoscopic cholecystectomy for biliary stone disease in the pediatric population has been well proven as the standard of care, similar to adult patients (18). ERCP was commonly applied from 1970 to 1979 to diagnose and treat hepatobiliary diseases in children. However, its use has been more restricted in recent years due to being an invasive procedure (19). For instance, Felux et al. (19) and Lou et al. (20) have reported that ERCP in children accounts for almost 3.3 and 4% of all ERCP procedures in their centers, respectively. In Asian countries, only a few studies with small sample sizes regarding pediatric ERCP have been performed (21). The success rate of endoscopic procedures, especially in children, requires a complete evaluation of the condition before ERCP, and it highly depends on the specialist’s skill. It is essential to understand that the ERCP indications should not be extended blindly because they may cause unnecessary complications (22).
To the best of our knowledge, no report on bile duct stone removal by ERCP in children has been published previously in Iran. This case is the first report of a successful pediatric ERCP for treating a bile duct obstruction due to a stone. The CBD stone was endoscopically removed. ERCP, along with other methods, can be considered a safe procedure for pediatric biliary diseases in well-equipped centers. The patient is still at risk for CBD stone formation because of having an anomaly in the biliary system. Laparoscopic cholecystectomy is necessary in this patient.