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Indian Pediatr 2018;55:216-218 |
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Etiology
and Outcome of Cholelithiasis in Turkish Children
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Masallah Baran 1,
Yeliz Cagan Appak2,
Gokhan Tumgor3,
Miray Karakoyun2,
Tunc Ozdemir2 and
Gokhan Koyluoglu1
From 1Department of Pediatric
Gastroenterology, Hepatology and Nutrition and Pediatric Surgery, Izmir
Katip Celebi University, Izmir; 2Department of Pediatric
Gastroenterology, Hepatology, Nutrition and Pediatric Surgery, SBU
Tepecik Training and Research Hospital, Izmir; and 3Department
of Pediatric Gastroenterology, Hepatology and Nutrition, Cukurova
University, Adana; Turkey.
Correspondence to: Yeliz Cagan Appak, Department of
Pediatric Gastroenterology Hepatology and Nutrition, Tepecik Training
and Research Hospital, Izmir, Turkey. Email:
[email protected]
Received: January 08, 2017;
Initial review: April 11, 2017;
Accepted: November 30, 2017.
Published online: December 14, 2017.
PII:S097475591600102
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Objective: The aim of this study was to examine
the etiology of gallstones in children and responses to ursodeoxycholic
acid (UDCA) treatment. Methods: 74 children with cholelithiasis
were recruited, and underwent ultrasonography to detect gallstones. All
relevant clinical information was recorded in a structured proforma.
Results: The commonest risk factor was a family history of
gallstones. Most children responded to UDCA treatment in the first six
months; children with hemolytic diseases showed no response to UDCA.
Conclusion: UDCA treatment may be useful before surgery in
asymptomatic patients of cholelithiasis without hemolytic diseases.
Keywords: Cholecystectomy, Gallstone, Ursodeoxycholic acid.
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The reported incidence of gallstones and bile sludge
in children is 1.9% and 1.46%, respectively [1]. In symptomatic
patients, a cholecystectomy is performed. Ursodeoxy-cholic acid (UDCA)
treatment dissolves gallstones in 19–37% of pediatric cases [2]. There
is no consensus on the most appropriate medical or surgical treatment of
pediatric gallstones.
This study investigated the demographic
characteristics and symptoms of children with gallstones, underlying
etiology, UDCA treatment response and cholecystectomy rates in two
hospitals in Turkey.
Methods
This was a retrospective study of children in whom
gallstones were determined at an outpatient gastroenterology clinic
between September 2009 and May 2016. Gallstones were detected by
abdominal ultrasonography (USG) and separated into five groups according
to the USG analysis: a large single stone
³1 cm, more than one
gallstone, multiple millimeter-sized stones, bile sludge and
microlithiasis (<3 mm) [3].
Data were collected on demographic characteristics,
personal and family histories, underlying disease that might lead to
stones, symptoms and laboratory findings. All the patients received UDCA
treatment at a dose of 20 mg/kg per day at admission. The UDCA treatment
was stopped in case of no treatment response after six months and in
cases of failure to achieve complete dissolution by one year [4].
Treatment was continued in cases where gallstones were partially
dissolved after six months [4]. A treatment response was considered
complete dissolution of gallstones, as determined by USG. The patients
underwent USG and laboratory examinations every three months. Ethical
approval for the study was obtained from the ethics committee.
Chi square test, Student’s t test and
Mann–Whitney U tests were conducted for comparisons of variables
using SPSS 19.0. Statistical significance was set at 0.05 in all tests.
Results
There were 74 children (33 males, aged 2 mo-17 y)
with mean (SD) age of 7.5 (4.3) y. Clinical findings on admission
included abdominal pain (38, 51.4%), nausea (25, 33.8%), vomiting (21,
28.4%), lack of appetite (15, 20.3%), cholestasis (3, 4.1%), and acholic
stool (1, 1.4%). Dehydration, acute pancreatitis and elevated
transaminase levels were observed in 4 (5.4%), 2 (2.7%) and 10 (13.5%)
cases, respectively. Asymptomatic gallstones were present in 29 (32.2%).
Risk factors and underlying diseases are shown in
Table I. The mean (SD) follow-up duration was 17 (17.1)
months, and duration of UDCA treatment was 9.7 (7.2) months (2–24
months). Gallstones disappeared within six months after treatment in 22
(29.7%) cases, and in 7 more by the end of 1.5 years. No change was
observed in 45 (60.8%) cases. The average time to the resolution of
gallstones was 4.9 (3.3) months. Adverse reactions (vomiting and
abdominal pain) occurred in only one patient (1.4%).
TABLE I Risk Factors in Turkish Children With Gallstones (N=74)
Risk factors |
n (%) |
Total parenteral nutrition |
13 (17.6) |
Family history |
11 (14.9) |
*Blood diseases |
6 (8.1) |
Prematurity |
5 (6.8) |
Oncological disease |
4 (5.4) |
Familial hyperlipidemia |
3 (4.1) |
Ceftriaxone use |
3 (4.1) |
Choledochal cyst |
3 (4.1) |
#Others |
5 (6.8) |
Idiopathic |
32 (43.2) |
*Thalessemia major and spherocytosis 2 each, 1 with
hemophilia;
#2 with obesity and 1 each with sepsis, trauma and cystic
fibrosis. |
A cholecystectomy was performed in 21 (28.4%) cases,
mostly laparoscopically, except for patients with choledochal cysts.
Thirteen of the patients were asymptomatic. Of these, eight patients had
a >1 cm gallstone, and five had multiple stones. Details of
response to treatment are shown in Table II.
TABLE II Treatment Response in Turkish Children with Cholelithiasis (N=74)
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Ursodeoxycholic Acid Treatment |
Characteristic |
Received |
duration (mo) |
Responded |
Underwent |
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n (%) |
mean (SD) |
n (%) |
cholecystectomy n (%) |
Size of gallstones |
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|
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Larger than 1 cm |
24(32.4) |
10.2(7.9) |
5(20.8) |
8(33.3) |
Multiple gallstones |
25(33.8) |
11.7(7.4) |
7(28) |
8(32) |
Multiple millimetre-sized gallstones |
17(23) |
7.5(4.7) |
10(58.8) |
4(23.5) |
Bile sludge |
5(6.8) |
3.6(1.5) |
5(100) |
None |
Microlithiasis |
3(4.1) |
11.6(10.9) |
2(66.7) |
1(66.6) |
Underlying disease |
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|
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Haematological disease |
6(8.1) |
7.4(3.9) |
1(16.7) |
1(16.7) |
Choledochal cyst |
3(4.1) |
6(5.1) |
None |
3(100) |
Drugs |
4(5.4) |
10.7(3.7) |
2(50) |
2(50) |
Prematurity |
5(6.7) |
14(9.1) |
None |
2(40) |
Hypercholesterolemia |
3(4.1) |
20(6.9) |
None |
3(100) |
Total parenteral nutrition |
13(17.6) |
10.5(8.3) |
3(23.1) |
5(38.5) |
Idiopathic |
32(43.2) |
9.9(6.9) |
14(43.8) |
7(21.9) |
Discussion
Hematological diseases are reported to be the most
frequent risk factor for gallstones, with a reported incidence of
8.9-50% [5,6]. In this study, a cholecystectomy was performed in 21
(28.4%) cases, most of whom were asymptomatic and had gallstones larger
than 1 cm. These findings are similar to that in the literature [7].
Some previous studies reported that UDCA had no
effect on gallstones, whereas others reported that it had some effect
[8]. Bogue, et al. [9] reported that an asymptomatic child could
be treated safely without surgical interventions. Interestingly, in the
present study, the gallstones were resolved in 20% of patients with
larger stones (>1 cm). Although no stone analysis was conducted, these
were most likely cholesterol stones, as these are the most common stones
found in children without haematological diseases [10]. The lack of
treatment response among the children with hemolytic diseases shows that
black pigment stones are resistant to UDCA treatment.
There is no consensus on the indications for a
cholecystectomy in asymptomatic pediatric patients [11]. A previous
study reported longer operative times and post-operative stays, in
addition to higher morbidity rates, among symptomatic patients who
underwent a cholecystectomy than among asymptomatic patients [12]. Thus,
as reported earlier, pediatric patients with gallstones who fail to
respond to medical treatment should undergo a cholecystectomy, performed
laparoscopically [13].
The present study has limitations common to
retrospective studies. A controlled prospective study with larger
numbers of patients is needed to provide additional evidence on the use
of UDCA treatment for gallstones in children.
In conclusion, whether medical or surgical treatment
should be recommended for gallstones remains a matter of debate,
especially in pediatric patients. UDCA can be recommended before
surgery, especially in asymptomatic patients without hemolytic diseases.
Contributors: All authors have contributed,
designed and approved the study.
Funding: None. Competing interest: None
stated.
What This Study Adds?
• Most children with cholelithiasis experience a response to
UDCA in the first six months, except those with hemolytic
diseases.
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