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\begin{document}

\title{Stone removal in a five-year-old child with extrahepatic biliary
obstruction using ERCP: A case report and a mini-review}



\author[1]{Ramin  Niknam}%
\author[1]{Seyede Maryam  Mahdavi Mortazavi}%
\author[1]{Mehdi Ghaderian Jahromi}%
\author[1]{Marzieh  Davoodi}%
\author[2]{Marzieh  Soheili}%
\author[1]{Maryam  Ataollahi}%
\author[1]{Reza Moshfeghinia}%
\affil[1]{Shiraz University of Medical Sciences}%
\affil[2]{Kermanshah University of Medical Sciences}%


\vspace{-1em}



  
  \date{May 05, 2024}


\begingroup
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\selectlanguage{english}
\begin{abstract}
We describe a 5-year-old child with extrahepatic biliary stone who
successfully underwent endoscopic retrograde cholangiopancreatography
for stone removal. He suffered from persistent colicky abdominal pain
accompanied by fever that biliary stone confirmed him. ERCP along with
other methods, can be considered a safe procedure for managing BD in
children.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Biliary obstruction is defined as the blockage of the extrahepatic
biliary system (1). The etiology of the benign or malignant extrahepatic
biliary system blockage involves various reasons, including gall stones
in the cystic duct causing pressure on the bile duct (Mirizzi syndrome),
choledochal cysts, and choledocholithiasis. Benign blockage of the
extrahepatic biliary system can be due to stricture diseases such as
fibrotic strictures from gall stone passage, PSC, and iatrogenic
strictures from bile duct cannulation. Neoplastic cases are presented
with stricture diseases causing biliary obstruction, including
pancreatic head cancer (causing distal CBD stricture), ampullary
carcinoma or adenoma, and cholangiocarcinoma (2). The most common cause
of biliary obstruction in developed countries is choledocholithiasis due
to cholesterol stones. Pigmented stones due to hemolysis and infectious
diseases, recurrent pyogenic cholangiohepatitis with increased risk for
cholangiocarcinoma, and calculi in intrahepatic bile ducts are common
etiologies in the Asian population which are rare in western countries.
Recurrent pyogenic cholangiohepatitis is characterized by recurrent
bacterial cholangitis, stricture, and dilatation of the biliary system.
Gallbladder malignancy is more common in East Asia, Central and South
America, Central and Eastern Europe, and the north of India (2, 3).

In general, choledocholithiasis is uncommon in children. Since the
extrahepatic biliary obstruction in children is very rare, most reports
describe the condition's etiology in adult patients. The prevalence of
cholelithiasis in pediatrics has been reported to be 0.13\% - 0.3\%.
However, the incidence is higher in obese children and adolescents and
is estimated at 2\% - 6.1\% (4).

More than 80\% to 90\% of all patients with CBD stones can be treated by
non-surgical methods through sphincterotomy and stone extraction in
combination with Dormia baskets or balloon catheters. In case of
non-extractable stones \textgreater{}1 cm, additional procedures such as
mechanical lithotripsy, including balloon dilatation, extracorporeal
shock-wave lithotripsy, electrohydraulic probe lithotripsy, laser
lithotripsy, stenting for immediate and definitive stone treatment are
applied (4). Endoscopic retrograde cholangiopancreatography is a
diagnostic and therapeutic technique routinely used for adults (5).
Relative to the published studies regarding adult ERCP, the articles on
pediatric ERCP remain limited for several reasons (6). Firstly, it is
technically more challenging to be used for children. Secondly,
pancreaticobiliary pathology in the pediatric population is rare, so the
study cannot have an adequate sample size. Additionally, in children
weighing more than 10 kg, pediatric ERCP duodenoscopes and accessories
have limited application (8). Moreover, the advancement of MRCP has
limited the use of ERCP for diagnosis.

This case report presents a CBD stone case with a left-sided gall
bladder treated with ERCP in an otherwise healthy 5-year-old male.

Left-sided gallbladder refers to a gallbladder located on the left side
of the ligamentum teres. It is a rare anomaly usually related to the
absence of segment IV, portal vein anomalies, or biliary system
anomalies. Diagnosis of the associated anomalies is essential for
managing liver transplantation, liver resection, and complicated
hepatolithiasis. Preoperative diagnosis of the left-sided gallbladder
with associated anomalies is required to reduce the risks of operative
complications (7).

\textbf{Case report}

A 5-year-old male was admitted to Namazi hospital (Shiraz, Iran),
suffering from a persistent colicky abdominal pain for the last 30 days.
Except for his premature birth, he had no history or family history of
the following conditions; previous medical problems, taking any
medications, past surgeries, abdominal trauma, weight loss, obesity,
metabolic syndrome, hemolytic diseases, severe skin itching (Bayler
disease), recurrent icterus, anemia, splenectomy, symptoms of chronic
liver disease or liver dysfunction, liver disorders (e.g., Wilson's
disease), steatorrhea, chronic diarrhea, as well as any underlying
causes of gallstone formation. The pain was localized in the epigastric
and periumbilical areas exacerbating after consuming dairy and high-fat
foods. The pain was accompanied by fever, vomiting, and constipation.
The patient was born with GA = 28W and a weight of 900 g. His current
body weight and height were 16 kg (10--25th percentile) and 112 cm
(50--75th percentile), respectively. Vital signs were stable; blood
pressure 100/70 mm Hg, heart rate 104 beats/min, respiratory rate 28
breaths/min, and body temperature of 36.5\selectlanguage{ngerman}°C. He had hepatomegaly and
abdominal tenderness in the epigastric and periumbilical areas in the
physical examination.

The hemoglobin, white blood cell, and platelet counts were 12.9 g/dL,
3700/mm3, and 192×106/mm3, respectively. Hemoglobin electrophoresis was
normal. Hemolysis was not noted. Blood chemistries were as following:
cholesterol 98 mg/dL (reference range, 120--200 mg/dL), total protein 6
mg/dL (6.1--7.9 mg/dL), albumin 4 mg/dL (3.5--5.6 mg/dL), alkaline
phosphatase 962 U/L , AST 104 U/L (15--40 U/L), ALT 180 U/L (5--45 U/L),
GGT 160U/L (5--32 U/L), total bilirubin 1.7 mg/dL (\textless{}2.0
mg/dL), amylase 44 U/L (16--91 U/L), and lipase 90 mg/dL (4--29 mg/dL)
(Table 1).

Abdominal ultrasound imaging revealed a distended gallbladder by diffuse
wall thickening with a maximum thickness of 3.5 mm, a CBD 10.8 mm in
diameter, and an 8.5-mm-sized stone in the distal CBD, suggestive of
acute cholecystitis.

Non per oral diet, intravenous hydration, and administration of
analgesics and antibiotics (cefotaxime and metronidazole) were started
after the patients' admission. The medical care team decided to perform
MRCP and ERCP to evaluate and manage the CBD stone. In MRCP evaluation,
mild dilation of central intrahepatic bile ducts and CBD (6 mm) was
apparent, associated with a dark signal of a 5 mm stone within the
pancreatic portion of CBD located at a 15 mm distance to the major
papilla. The gall bladder's position was on the left side of the
subhepatic area, concurrent with portal vein abnormality. The main
portal vein was trifurcated, and ascending portion (umbilical portion)
of the left portal vein was hypoplastic. Additionally, small teres
ligament differentiation of segment IV was suboptimal (Figure 1).

Since our center is well-equipped, ERCP was performed under general
anesthesia. During the procedure, dilatation of CBD with a stone in the
middle was investigated (Figure 2). After sphincterotomy, a 10*15 mm
pigmented stone was removed using a stone retrieval balloon (Figure 3).
After the stone removal, all signs and symptoms were alleviated. There
were neither any complications during ERCP nor after discharge. The
patient was discharged, and Ursodeoxycholic acid (10 mg/kg/dose) and
Polyethyenglycol syrup (PEG) (1 cc/kg/day) were prescribed for 20 days.
No complications, signs, or symptoms were observed in the follow-up
visit, and all lab results and abdominal sonography were within the
normal range. Finally, the patient was referred for elective
cholecystectomy.

Table 1: Laboratory data during hospitalization\selectlanguage{english}
\begin{longtable}[]{@{}lllll@{}}
\toprule
\begin{minipage}[b]{0.17\columnwidth}\raggedright\strut
4 months later\strut
\end{minipage} & \begin{minipage}[b]{0.17\columnwidth}\raggedright\strut
12 Days After Admission\strut
\end{minipage} & \begin{minipage}[b]{0.17\columnwidth}\raggedright\strut
3 Day After Admission\strut
\end{minipage} & \begin{minipage}[b]{0.17\columnwidth}\raggedright\strut
On Admission\strut
\end{minipage} & \begin{minipage}[b]{0.17\columnwidth}\raggedright\strut
Lab Data\strut
\end{minipage}\tabularnewline
\midrule
\endhead
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
6490\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
7600\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
3700\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{WBC, /mm\textsuperscript{3}}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
13/9\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
12/1\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
12/9\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Hb, gm/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
299\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
467000\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
192000\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Platelet, /mm\textsuperscript{3}}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
14\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{PT, sec}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
1/1\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{INR, index}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
34\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{PTT, sec}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{B.U.N, mg/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
0/37\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Cr, mg/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
135\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Na, mEq/L}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
3/7\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{K, mEq/L}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
25\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
64\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
104\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{AST, IU/L}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
56\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
131\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
180\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{ALT, IU/L}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
406\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
665\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
1037\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
962\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{ALK.P, IU/L}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
9/4\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
114\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
225\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
160\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{GGT}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
1/2\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
1/7\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Total bilirubin, mg/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
0/5\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
0/5\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Direct bilirubin, mg/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
6/4\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
6\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Total protein, g/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
4/2\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
4\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Albumin, g/dL}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
Neg\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Coombs}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
44\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Amylase}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
90\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{lipase}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
12\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{ESR, mm/h}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
3/6\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
2\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{CRP, mg/L}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
98\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{cholesterol}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
122\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
triglyceride\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.19\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.19\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.19\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.19\columnwidth}\raggedright\strut
Neg\strut
\end{minipage} & \begin{minipage}[t]{0.19\columnwidth}\raggedright\strut
Viral marker (HBS Ag, HAV Ig M, HCV Ab)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
437\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
135\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Urine COPPER}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
Neg\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{IgG}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
Neg\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Anti-LKM}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
Neg\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{ANA}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
363\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{LDH}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
3/1\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Uric acid}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
No growth\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Blood culture}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
No growth\strut
\end{minipage} & \begin{minipage}[t]{0.17\columnwidth}\raggedright\strut
\textbf{Urine culture}\strut
\end{minipage}\tabularnewline
\bottomrule
\end{longtable}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

\end{center}
\end{figure}

Figure 1: A) axial T2 MRCP sequence: gall bladder in left subhepatic
area. B) axial T2 MRCP sequence: tiny signal void (dark) stone in the
pancreatic portion of CBD. C) coronal T2 MRCP sequence: signal void tiny
stone in the distal part of the CBD. D) coronal 3D reconstruction MRCP
image presenting tiny stone in CBD.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image2/image2}

\end{center}
\end{figure}

Figure 2: ERCP: dilatation of the CBD with a filling defect (left image)
in the middle part and stone removal using a stone retrieval balloon
(middle and right images).\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image3/image3}

\end{center}
\end{figure}

Figure 3: Endoscopic view of pigmented stone after removal using a stone
retrieval balloon

\textbf{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.

\textbf{Availability of data and materials}

The data supporting this case report's findings is available from the
corresponding author upon request.

\textbf{Abbreviations}

PSC: primary sclerosing cholangitis, CBD: common bile duct, ERCP:
Endoscopic retrograde cholangiopancreatography, GA: gestational age,
AST: aspartate aminotransferase, ALT: alanine aminotransferase, GGT:
gamma-glutamyl transpeptidase, MRCP: magnetic resonance
cholangiopancreatography, MRI: magnetic resonance imaging, PEG:
polyethylene glycol, PFIC: (progressive familial intrahepatic
cholestasis), NEC: necrotizing enterocolitis

\textbf{Declarations}

\textbf{Ethics approval and consent to participate}

This research was approved by the ethics committee of shiraz university
of medical sciences with IR.SUMS.REC.1401.665 code.

\textbf{Consent for publication}

Written informed consent was obtained from the patient's mother to
publish this case report and any accompanying images. All authors have
viewed and agreed to the submission.

\textbf{Competing interests}

All authors declare that this manuscript has no conflict of interest.

\textbf{Funding}

Not applicable

\textbf{Authors' contributions}

RN and SMMM performed the endoscopy and cared for the patient. MS, MD
and RM collected the clinical data and wrote the manuscript. MGHJ helped
with the imaging diagnosis. MA revised the manuscript. All authors have
read and approved the final manuscript.

\textbf{Acknowledgments}

Not applicable.

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\selectlanguage{english}
\FloatBarrier
\end{document}

