Cholecystectomy Versus Cholecystolithotomy for Cholelithiasis in Childhood: Long-Term Outcome By Diane De Caluwe´, Usama Akl, and Martin Corbally Dublin, Ireland
Background/Purpose: The presence of cholelithiasis is being reported with increased frequency in childhood. Little is known about the natural history of the disease, and only a few studies have been published regarding long-term results of treated patients. Controversy still exists regarding optimal treatment. Both cholecystectomy and cholecystolithotomy with gallbladder preservation have been recommended as the preferred operative intervention. The purpose of this study was to compare the long-term outcome of cholecystectomy versus cholecystolithotomy for symptomatic gallbladder disease in children. Methods: The charts of all patients with symptomatic cholelithiasis treated in the Dublin Paediatric Hospitals during a 25-year period from 1974 till 1999 were reviewed. Data obtained included age, sex, age at presentation of symptoms, methods of diagnosis, indications for operative treatment, time interval between presentation of symptoms and surgery, surgical technique, performance of a preoperative or intraoperative cholangiogram, stone biochemistry, gallbladder histology, radiologic follow-up, the presence of recurrent or residual stones and symptoms, and the need for reoperation. Patient data were grouped according to method of surgery. All parameters were compared and evaluated. Fol-
low-up was by way of telephone contact with all patients and completion of a questionnaire.
Results: There were 18 patients over a 25-year period. Eight patients underwent cholecystectomy, and 10 patients had a cholecystolithotomy. Median follow-up was 2 years in the cholecystectomy group and 5 years in the cholecystolithotomy group. All patients in the cholecystectomy group are asymptomatic and have no recurrent or residual stones on follow-up ultrasound scan. Thirty percent of the patients in the cholecystolithotomy group have recurrent right upper quadrant pain, and 30% show recurrent stones 9.5 months (range, 7 to 12 months) postoperatively. One patient underwent cholecystectomy 8.5 months postcholecystolithotomy. Conclusions: The symptomatic high stone recurrence rate postcholecystolithotomy seen in our series suggest that cholecystectomy is the preferred treatment in patients with symptomatic gallbladder disease. J Pediatr Surg 36:1518-1521. Copyright © 2001 by W.B. Saunders Company. INDEX WORDS: Cholelithiasis, cholecystectomy, cholecystolithotomy.
HOLELITHIASIS is a well established disorder in the pediatric age group and must be considered in the differential diagnosis of recurrent abdominal pain in children.1 Although uncommon during the first year of life, it is being diagnosed with increasing frequency in neonates and infants, probably because of the widespread use of ultrasonography.2-5 Recent reports indicate also a rising incidence of gallbladder disease among older children and adolescents.6 Infants, children, and adolescents with cholelithiasis constitute 3 different populations in relation to pathogenesis, predisposing factors, symptomatology, and outcome.2,3,5-7 There is still controversy regarding the optimum treatment of cholelithiasis in
C
children.4,5,8 Spontaneous elimination or resolution of calculi may occur.4,5,9,10 Some investigators consider cholecystectomy the treatment of choice, whereas others recommend cholecystolithotomy.1-3,5,8,9,11,12 Recurrent and residual gallstones postcholecystolithotomy have been shown in a number of reports.8,13,14 However, an increased risk of right-sided colonic cancer has been suggested after cholecystectomy.5,8,15 Only a few studies have been published regarding long-term results of cholecystectomy and cholecystolithotomy in childhood.8 The aim of this study is to compare the long-term outcome of cholecystectomy versus cholecystolithotomy in the treatment of cholelithiasis in the pediatric age group.
From the Department of Pediatric Surgery and Children’s Research Centre, Our Lady’s Hospital for Sick Children, Dublin, Ireland. Address reprint requests to Diane De Caluwe´, Children’s Research Centre, Our Lady’s Hospital for Sick Children, Crumlin, Dublin 12, Ireland. Copyright © 2001 by W.B. Saunders Company 0022-3468/01/3610-0010$35.00/0 doi:10.1053/jpsu.2001.27035
A retrospective study of the medical records of all children 15 years or younger admitted to the 3 pediatric hospitals in Dublin with documented symptomatic cholelithiasis from January 1, 1974 to December 31, 1999 was performed. Charts were identified by reviewing the logs of the pediatric surgical service and by conducting a computerized institutional chart search within the hospitals. Data obtained included age, sex, age at presentation of signs and symptoms, methods of diagnosis, indications for operative treatment, time interval between presentation of symptoms and surgery, surgical technique, performance
MATERIALS AND METHODS
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Journal of Pediatric Surgery, Vol 36, No 10 (October), 2001: pp 1518-1521
CHOLECYSTECTOMY VERSUS CHOLECYSTOLITHOTOMY
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Table 1. Profile of Eight Patients With Cholelithiasis Who Underwent Cholecystectomy Clinic Follow-Up
Radiology Follow-Up
Well Well Well
No No US at 11 mo: no stones
No No No
2 yr 3.5 yr 1.5 yr
Well
US at 4.5 mo no stones
No
1.5 yr
US US, CT, MRI US
Recurrent pain, vomiting Recurrent cholecystitis Obstructive cholangitis, pancreatitis Recurrent cholecystitis, vomiting Recurrent pain, jaundice Pancreatitis Recurrent pain, vomiting
Well Well Well
US at 10 mo: no stones US at 5 mo: no stones US at 2 mo: no stones
US, cholecystogram
Recurrent pain
Well
No
No No Incisional hernia No
Patient
Age and Sex
Diagnosis
Indication for Surgery
1 2 3
15 yr, F 23 yr, F 13 yr, M
PFA US, cholecystogram US, MRCP
4
16 yr, F
US, CT
5 6 7
6 yr, M 14 yr, F 9 yr, F
8
23 yr, F
Reoperation
Follow-Up
10 mo 5 mo 4 yr 9.5 yr
Abbreviations: PFA, plain abdominal film; US, ultrasound scan; MRCP, magnetic resonance cholangiopancreaticography.
formed (Table 2). Five patients in this group had an oral cholangiogram preoperatively; no patient had an intraoperative cholangiogram. One patient in this group initially underwent extracorporal shock wave lithotripsy (ESWL) leaving him with a number of small fragmented stones, and cholecystolithotomy was performed 1 year later. Indications for surgery in the 2 groups were recurrent episodes of right upper quadrant (RUQ) pain associated with recurrent cholelithiasis in 89% of the patients, obstructive jaundice in 2 patients, and a nonfunctioning gallbladder with multiple stones in one patient. Presenting symptoms were recurrent RUQ pain, anorexia, nausea, vomiting, obstructive jaundice, and pancreatitis. The median age at presentation of symptoms was 8.5 years (range, 2 days to 18 years). Diagnosis was made by an abdominal ultrasound scan in 83% and a PFA in 17%. Additional x-rays done were oral cholecystogram (33%), HIDA scan (6%), percutaneous cholecystogram (6%), MRCP (6%), computed tomography (CT; 11%), magnetic resonance imaging (MRI; 6%), and intravenous cholangiogram (6%). Median time episode
of a preoperative or intraoperative cholangiogram, stone biochemistry, gallbladder histology, radiologic follow-up, the presence of recurrent or residual stones and symptoms, and the need for reoperation. Patient data were grouped according method of surgery. Group 1 consisted of patients who underwent cholecystectomy, and group 2 of patients who underwent cholecystotomy with removal of the stone only. All parameters were compared and evaluated. A telephone interview was obtained with all patients for the purpose of follow-up.
RESULTS
Eighteen patients were treated over a 25-year period. The male to female ratio was 6:12. The median age for the boys was 12 years (range, 5 to 21 years) and 15 years (range, 9 to 23 years) for the girls. Eight patients underwent cholecystectomy, 6 by open surgery and 2 by laparoscopy (Table 1). Five of these patients underwent intraoperative cholangiogram and one a preoperative oral cholangiogram. One patient in this group who had gallbladder stones and 2 large stones in the common bile duct (CBD) initially underwent endoscopic retrograde cholangiopancreaticography (ERCP) and a sphincterotomy. Three weeks later, an open cholecystectomy was carried out. Ten patients had a cholecystolithotomy per-
Table 2. Profile of 10 Patients With Cholelithiasis Who Underwent Cholecystolithotomy Patient
Age, Sex
Diagnosis
1 2 3
15 yr, F 18 yr, F 11 yr, M
4 5
12 yr, F 3.5 yr, M
US PFA, US, oral cholecystogram US, HIDA, percutaneous cholangiogram PFA US
6 7 8 9
22 21 11 17
PFA, oral cholecystogram US US, PFA US, oral cholecystogram
10
yr, yr, yr, yr,
F M F M
15 yr, F
PFA, US, IV cholangiogram, oral cholecystogram
Indication for Surgery
Recurrent pain Recurrent pain Obstructive jaundice Recurrent pain Recurrent pain, vomiting Recurrent pain Recurrent pain Recurrent pain Incidental finding Recurrent pain, vomiting
Abbreviation: PFA, plain abdominal film; US, ultrasound scan.
Clinic Follow-Up
Radiology Follow-Up
Recurrent pain at 7 mo Recurrent stones Well US Recurrent pain Recurrent stones
Reoperation
Follow-Up
Cholecystectomy No No
10 mo 5 yr 9 yr
Well Well
US US
No No
3 yr 3 yr
Well Recurrent pain Well Well
Recurrent stones US US US
No No No No
8 yr 6 yr 19 mo 5 yr
Well
US, oral cholecystogram
No
6 yr
DE CALUWE´, AKL, AND CORBALLY
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between presentation of symptoms and surgery was 2 months (range, 1 month to 10 years). Histology of the gallbladder wall showed chronic cholecystitis in 3, mild inflammation in 3, and no inflammation in 1 of the sent specimens in the cholecystectomy group and mild inflammation in 2 and chronic cholecystitis in 2 in the cholecystolithotomy group. Histology of the stones showed 5 pigment stones, 2 calcium stones, and one cholesterol stone. Median follow-up was 2 years (range, 6 months to 9.5 years) in the cholecystectomy group and 5 years (range, 8 months to 9.5 years) in the cholecystolithotomy group. All patients in the cholecystectomy group are asymptomatic and have no recurrent or residual stones on follow-up ultrasound scan. One patient in this group underwent surgery for an incisional hernia. Thirty percent of the patients in the cholecystolithotomy group show recurrent stones on follow-up ultrasound scan 9.5 months (range, 7 to 12 months) postoperatively, and 30% have recurrent RUQ pain. One patient, therefore, underwent cholecystectomy 8.5 months postcholecystolithotomy. Initial stone biochemistry showed pigment stones in these patients. Three patients had multiple recurrent stones on ultrasound scan. DISCUSSION
Cholelithiasis is being reported with increasing frequency in the pediatric age group.4,6 The natural history of cholelithiasis in childhood still remains unclear, and most series are small, and few have reported detailed long-term follow-up of treated patients.6,8,10,11 Controversy exists as to the optimal management of cholelithiasis in children and varies between cholecystectomy or cholecystostomy with stone removal and gallbladder preservation.4,5,8 Both surgery and interventional radiologic procedures have been advocated, but also nonoperative management of gallstones has been reported.4,8 Spontaneous resolution or elimination of gallbladder sludge or gallstones has been documented in neonates and young infants, probably because of stone migration through the common bile duct.4,5,9,10 Conservative management is suggested in the asymptomatic child with no biochemical or radiologic signs of common bile duct obstruction with yearly follow-up ultrasonography. However, some series have a policy to recommend surgery in
these children to rule out the risk of symptoms developing or the sequelae of gallstones.3,4,8 Operative intervention is recommended for symptomatic patients and those with calcified stones because these are unlikely to resolve spontaneously.2,5,9 Cholecystectomy is regarded by many as the treatment of choice, especially in cases of congenital anomalies of the biliary tree or nonfunctioning gallbladders on cholecystography.5,11 Other investigators have recommended cholecystolithotomy with preservation of the gallbladder in the absence of inflammatory changes.1,3,5,8,9,11 This, however, carries the risk of residual or recurrent cholelithiasis.13 Thirty percent of the patients in our series had recurrent stones within a year postoperatively. The initially removed stones in these patients were pigment stones. Thirty percent of the patients were symptomatic with a median follow-up of 5 years. We consider this as unacceptable. Cholecystectomy helps avoid the possibility of residual or recurrent gallstones but does not prevent recurrent choledocholithiasis.5,8 However, none of the patients in our series presented with recurrent stones or choledocholithiasis on follow-up ultrasound scan. In addition, it has been suggested that changes in bile salt mechanism that occur with the continuous secretion of bile into the gut after cholecystectomy may be associated with an increased risk of right-sided colonic cancer.5,8,15 This continuous secretion leads to enhanced formation of secondary bile acids, such as lithocholic and deoxycholic acid, because of increased enterohepatic circulation and degradation of primary bile acids by intestinal bacteria. These secondary bile acids could act as cocarcinogens in adults.15 If any association would exist, cholecystectomy in childhood would be undesirable and should be carried out with some reservation.8 However, it also is proposed that much or all of the association observed in some investigations could be an artefact caused by a heightened intensity of diagnostic and therapeutic activity for mild abdominal symptoms in adults.15 In our series, we have not noticed any association with any form of colon cancer in children postcholecystectomy. The symptomatic high stone recurrence rate after cholecystolithotomy noticed in our series suggests that cholecystectomy is the preferred treatment in patients with symptomatic gallbladder disease.
REFERENCES 1. Moossa AR: Cholelithiasis in childhood. JR Coll Surg Edinb 18:42-46, 1973 2. Schirmer WJ, Grisoni ER, Gaudener MWL: The spectrum of cholelithiasis in the first year of life. J Pediatr Surg 24:1064-1067, 1989 3. Jacir NN, Anderson KD, Eichelberger M, et al: Cholelithiasis in infancy: Resolution of gallstones in three of four infants. J Pediatr Surg 21:567-569, 1986
4. Debray D, Pariente D, Gauthier F, et al: Cholelithiasis in infancy: A study of 40 cases. J Pediatr 122:385-391, 1992 5. Asabe K, Handa N: Infant Cholelithiasis: Report of a Case. Surg Today 27:71-75, 1997 6. Fisher M, Rosenstein J, Schussheim A, et al: Gallbladder disease in children and adolescents. J Adol Health Care 1:309-312, 1981 7. Pokorny WJ, Saleem M, O’Gorman RB, et al: Cholelithiasis and cholecystitis in childhood. Am J Surg 148:742-744, 1984
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8. Robertson JFR, Carachi R, Sweet EM, et al: Cholelithiasis in childhood: A follow-up study. J Pediatr Surg 23:246-249, 1988 9. Grosfeld JL, Rescorla FJ, Skinner MA, et al: The spectrum of biliary tract disorders in infants and children. Arch Surg 129:513-520, 1994 10. Bohle AS, Grimm H, Mengel W: Case report: Cholelithiasis with common bile duct obstruction in a 20-week-old infant. Eur J Pediatr Surg 5:57-58, 1995 11. O’Donnell B, Puri P: Long-term results of simple removal of pigment gallstones in childhood. Prog Pediatr Surg 10:121-127, 1977
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12. Holcomb GW Jr, O’Neill JA, Holcomb GW: Cholecystitis, cholelithiasis and common duct stenosis in children and adolescents. Ann Surg 191:626-635, 1980 13. Gibney RG, Chow K, So CB, et al: Gallstone recurrence after cholecystolithotomy. AJR 153:287-289, 1989 14. Norrby S, Schonebeck J: Long-term results with cholecystolithotomy. Acta Chir Scand 136:711-713, 1970 15. Friedman GD, Goldhaber MK, Quesenberry CP Jr: Cholecystectomy and large bowel cancer. Lancet 18:906-908, 1987