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.