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 Table of Contents  
ORIGINAL RESEARCH ARTICLE
Year : 2020  |  Volume : 9  |  Issue : 2  |  Page : 68-73

Pediatric gallstone disease - Experience of two surgeons


1 Department of Pediatric Surgery, IMS and SUM Hospital, Bhubaneswar, Odisha, India
2 Department of General Surgery, VCSGGMS and RI (HNB Base Hospital), Pauri Garhwal, Uttarakhand, India
3 Executive Director, GRID Council (Generating Research Insights for Development), NCR, Delhi, India
4 Dental Surgeon/Consultant, Biostatistician, DPDOCC, Bareilly, Uttar Pradesh, India

Date of Submission16-Aug-2020
Date of Acceptance22-Sep-2020
Date of Web Publication27-Apr-2021

Correspondence Address:
Dr. Keshri Amit
Department of General Surgery, VCSGGMS and RI (HNB Base Hospital), Srikot, Srinagar, Pauri Garhwal, Uttarakhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpai.jpai_5_20

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  Abstract 


Introduction: Gallstone disease is being increasingly diagnosed as a cause of pediatric pain abdomen, requiring cholecystectomy. Most cases are idiopathic, while uncommon etiologic associations nowadays include obesity and hemolytic anemias/hemoglobinopathies; prolonged disease and high fat diet are predisposing factors. Management aspect is similar to adults. Aim: This study aims to compare the profile, interventions, and outcomes of pediatric gallstone disease patients, having undergone cholecystectomy, between two surgeons working at separate centers in India. Materials and Methods: Case records of all pediatric patients (≤15 years' age) having undergone cholecystectomy at both the centers by the respective surgeons (AP = 43 and KA = 17; total = 60 cases), between August 2015 and November 2019, were examined. Data were tabulated and inferences were drawn. Results: Male patients outnumbered female patients in both the studies (AP – M: F = 24:19, KA – M: F = 12:5). Anemia/hypoproteinemia and obesity were prevalent among the operated children (AP = 6 and KA = 3). Biliary pancreatitis (AP = 2 and KA = 2)/choledocholithiasis (AP = 7 and KA = 3) was the uncommon presentation; common bile duct obstruction with cholangitis/sepsis was rarer still. Laparoscopic cholecystectomy was the commonly performed procedure (AP lap: open = 41:2 and KA lap: open = 10:7). Choledocholithiasis patients underwent prior endoscopic retrograde cholangio-pancreatography (AP = 7 and KA = 3). Most patients had cholesterol/mixed gallstones. Complications were minimal, comparable with adult procedures. Conclusion: Pediatric gallstone disease is understated in literature and underestimated in clinical practice. Early diagnosis and timely referral to a surgeon for appropriate treatment can help prevent complications and reduce time–work–loss to child/parent.

Keywords: Cholecystectomy, cholelithiasis, laparoscopic cholecystectomy, pain abdomen, pediatric gallstone disease


How to cite this article:
Pradhan A, Amit K, Mohapatra A, Punita K. Pediatric gallstone disease - Experience of two surgeons. J Pediatr Assoc India 2020;9:68-73

How to cite this URL:
Pradhan A, Amit K, Mohapatra A, Punita K. Pediatric gallstone disease - Experience of two surgeons. J Pediatr Assoc India [serial online] 2020 [cited 2022 Jul 3];9:68-73. Available from: http://www.jpai.in//text.asp?2020/9/2/68/314818




  Introduction Top


Cholelithiasis has an incidence rate of ~0.15%–0.22% in the pediatric population, and rising, with female adolescents affected more commonly.[1] The cause of calculous cholecystitis in pediatric patients is idiopathic nowadays world over; uncommon causes and other high-risk groups include children with obesity, hemolytic anemias/hemoglobinopathies, ileal resection/disease, prolonged disease/total parenteral nutrition, certain drugs (ceftriaxone, furosemide, octreotide, cyclosporin, etc.), rarely progressive familial intrahepatic cholestasis Type 3, and a high fat diet; patients having undergone previous surgery are also predisposed to develop cholelithiasis.[1],[2],[3]

Presentation is with recurrent/acute pain in the upper abdomen, that may radiate to the right shoulder or dyspepsia, with a positive Murphy's sign. Icterus or pain radiating to the back suggests stone in the common bile duct (CBD) or ampulla, causing pancreatitis. Ultrasonography (USG)/(magnetic resonance imaging ± magnetic resonance cholangio-pancreatography) abdomen usually forms the first line of diagnosis, aided by Liver / Kidney Function Tests (LKFT)/complete blood count and other relevant investigations. Specific blood/hemoglobin analytic tests may be required to diagnose hemolytic disorders/hemoglobinopathies. Some of these patients might have been initially treated with Urso-Deoxy Cholic Acid (UDCA) and/or antibiotics/analgesics.[1],[2],[3],[4]

Cholangitis may occur with a gallstone blocking the CBD.  Escherichia More Details coli and Klebsiella, Pseudomonas, and Enterococcus species are the commonly involved organisms. Choledocholithiasis occurs in ~ 11% of children with cholelithiasis and ~ 20% of pediatric patients with gallstone pancreatitis, caused by the passage of stones through the cystic duct with entrapment at the papilla of Vater.[1],[2],[3],[4]

Laparoscopic cholecystectomy is the procedure of choice for cholelithiasis/gallbladder pathology; open procedure is usually resorted to only in the presence of choledocholithiasis or some complications or variable/unclear anatomy.[5] Most calculi are cholesterol/mixed type, and rarely there are pigment stones. Prognosis is excellent in pediatric patients treated timely with surgery, with an expected early recovery, while complication rate is at par with adult procedures. Postcholecystectomy syndrome is of concern in some patients. Dietary modifications in children could be advised.[1],[2],[3],[4],[5]


  Methods Top


Aim

We aim to outline the profile, interventions, and outcomes among pediatric patients (≤15 years' age) having undergone cholecystectomy at two tertiary care centers in India.

Study setting and timelines

We report data collected from two centers, that is, eastern India: IMS and SUM Hospital, Bhubaneswar, Odisha, and northern India: VCSGGMS and RI, Srinagar, Uttarakhand, by the respective surgeons (AP – pediatric surgeon; KA – general surgeon), between August 2015 and November 2019. Both surgeons have about 10 years of experience operating on pediatric cases.

Participants

Case records of all pediatric patients (≤15 years' age), referred or directly presenting to the surgeons, having undergone cholecystectomy for symptomatic cholelithiasis (AP = 43 and KA = 17; total = 60 cases), including those gallbladder calculus cases associated with choledocholithiasis/gallstone pancreatitis/other accompanying pathology, during the above period, were included in the study, while excluding those having asymptomatic gallstones, those managed medically/conservatively, those who had underwent incidental/prophylactic cholecystectomies, or those suffering from acalculous cholecystitis. Both laparoscopic and open approaches were taken into consideration; thothose patients suffering from choledocholithiasis underwent endoscopic retrograde cholangio-pancreatography (ERCP) and accompanying procedure (calculus extraction/stenting/sphincterotomy) prior to surgery; with open approach resorted to in such unsuccessful cases, and no laparoscopic CBD exploration done for any case of choledocholithiasis.

Standard four-port laparoscopic cholecystectomy technique was preferably performed in all cases, with 10–12 mmHg pressure for pneumoperitoneum, and extra port (s) was/were utilized in the cases of adhesions or difficult progress, as per the surgeon's preference. Conversion to open procedure was done in the cases of unmanageable dense adhesions/obscure anatomy, etc., The planned open procedures were performed with the standard Kocher's right subcostal incision, extended as per requirement. The excised specimen of the gallbladder/surrounding tissue was sent for histopathological examination (HPE).[5] ERAS protocols were preferably followed in all cases perioperatively.[6]

Appropriate informed and written consent was taken from the parents/patients included in this study. The operated cases were observed postoperatively and discharged as per regain of appropriate functional status and oral intake by the patients. Thereafter, they were recalled for outpatient department visit at 1 week for stapler/suture removal and again at the 3rd to 6th month from the date of operation for review, earlier if any further complaints arise, or later if needed. Case notes and follow-up records were made, and documentation was done and preserved.

Appropriate clearance was sought from the institutional ethical committees/institutional review boards. Google (Drive/Docs/Office Suite) (google.com/drive), LinuxMint (linuxmint.com), and LibreOffice (libreoffice.org) were used for data tabulation, analysis, manipulation, interpretation, graphing, and charting, with results as observation numbers/percentages, statistical analyses, and relevant comparisons. There were no conflict of interests and no source of support/funding. The content of this article was expressly written by the authors listed. No ghostwriters were used to write this article.


  Results Top


Pediatric patients (≤15 years' age) having undergone cholecystectomy, were included in the study by the above-outlined criteria; they numbered NAP= 43 and NKA= 17, respectively (Ntotal=60) among the two surgeons working separately at their respective centers [Table 1]. The age-wise distribution of the patients showed the “7–12-year” group to have the highest number of patients in both the studies (NAP[7–12 years] = 23 and NKA[7–12 years] = 10, overall N (7–12 year) = 33 [55.5%]), with a median age of 10 years. The number of male patients was higher across all groups (M: FAP = 24:19, M: FKA = 12:5, overall M: F = 36:24 [60% male patients]). While the cause of most of the cases of cholelithiasis was idiopathic (55; 91.7%), three of the cases were associated with obesity (body mass index ≥30 kg/m2) and one patient had β-thalassemia minor, while another had a rare “double” gallbladder type of congenital anomaly [Graph 1]. Anemia/malnutrition (hypoproteinemia) was found in nine cases (15.0%).
Table 1: Pediatric gallstone disease (August’ 15–November’ 19) – two surgeons/centers – study results

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Choledocholithiasis accompanying cholelithiasis was present in ten cases (10; 16.7%), of which nine underwent ERCP + procedure prior to the operative intervention. Overall, two patients presented with jaundice due to obstructed CBD, but none showed signs of cholangitis. Features of gallstone pancreatitis accompanying cholelithiasis, confirmed with serum lipase/amylase levels, were seen in four patients.

Laparoscopic cholecystectomy is the procedure of choice and the gold standard for treating gallstones; overall, among the total patients handled by both the surgeons, 49 patients underwent the laparoscopic cholecystectomy procedure (NAP (Lap-Ch) = 40 of 43, NKA (Lap-Ch) = 9 of 17), while conversion to an open procedure from laparoscopic attempt had to be done in one case each by both the surgeons, citing accessory cystic duct (of Luschka) by AP and difficult anatomy and adhesions by KA. Open cholecystectomy was planned and done in one case by AP and six cases by KA, in anticipation of adhesions/abscess and other complications due to delayed presentation by the patient; one case each was a planned open CBD exploration along with the cholecystectomy (for inadequate removal of calculi during the prior ERCP [AP] and no ERCP [KA]) [Graph 2].



Intraoperative drain insertion had to be done in four and three cases (AP: KA = 4:3; seven overall), to manage the residual pus/ooze from the gallbladder fossa/bed, while no other intra/postoperative complications were encountered in the cases. Minor infection (port-site / wound / surgical-site) occurred in one patient each among the procedures done by the two surgeons, which were adequately managed with antiseptic dressings. The mean postoperative duration of stay was about 1.5 days in case of laparoscopic and about 2.3 days for open surgery technique done among patients handled by both the surgeons, with patients discharged on short-course oral antibiotics (3–5 days), analgesics on SOS basis, and other supportive medications, with advice on minor dietary and activity restrictions.

HPE of the excised tissue specimens revealed chronic cholecystitis in most of the cases (calculous) (48; 80%), whereas others showed acute (3), acute-on-chronic (8), and gangrenous (1) features; the findings correlated well and were congruous with the patients' constellation of symptomatology. Upon visual inspection and palpation of the retrieved gallstones, the mixed type seemed to be the most common (86.7%), followed by pure cholesterol and pigment types, in both the studies [Graph 3].




  Discussion Top


Knowledge of the common causes of pediatric pain abdomen as well as etiology and presentation of cholelithiasis in the community is essential to practice nowadays. Parents of children with recurrent/acute pain in the upper abdomen usually seek pediatrician's advice, who after investigating and determining an operable cause refer them to their surgery colleagues. Laparoscopic cholecystectomy is the treatment of choice for cholelithiasis and is safe enough in expert hands to be considered for all age groups, with the open procedure having specific indications.[1],[2],[3],[4],[5] ERAS protocols promote early recovery and reduce perioperative stress while ensuring the best possible outcomes.[6]

Holcomb et al.[7] and Esposito et al.[8] have shown that laparoscopic cholecystectomy in indicated patients is a safe and effective strategy in the pediatric age group, as also the protocol followed by us [Graph 2]. Studies by Niyogi et al.[9] and Nachulewicz[10] and several similar recent studies depict a higher number of cholecystectomies being performed the world over nowadays, not only due to the higher detection rate of the pathology by better imaging and diagnostics but also due to an actual rise in the incidence of gallstones among various populations. The evidence-based demonstration of the safe outcomes of this procedure, with lesser complication rates in pediatric population than adults, shown by St Peter et al.[11] and others has led to the universal acceptance and adoption of this standard treatment modality. The recent changing trends and epidemiology mandate surgeons to seek the etiology of abdominal pain among their pediatric patients and operate at a lower threshold for gallbladder pathology, as shown by Novielloa et al.[12]

Kumar,[13] Bhasin et al.,[14] Jogendra,[15] Maharjan et al.[16] and many others have carried out recent studies on laparoscopic cholecystectomy in pediatric patients at their institutes, catering to the regional populations, performed by both specialist pediatric surgeons and general surgeons, with favorable outcomes. Although many of these compilations depict a higher percentage of female patients in their study, as against both our groups, this is subject to “admission bias” at all the centers. Idiopathic gallstones are the most common, while those related to obesity and lithogenic drugs are on the rise. Gallstones in relation to hemolytic disorders/hemoglobinopathies are seen less often, thereby implying that they are not as common as previously portrayed in medical literature [Graph 1] and [Table 1].

Patients in Holcomb et al.'s[7] study had a mean age of 105 months (<10 years); most of the patients underwent laparoscopic cholecystectomy and none had any significant complications. Esposito et al.'s[8] multi-institutional study had patients undergoing laparoscopic cholecystectomy; the median age of the patients was 8.5 years, with more females in the group and most were affected by idiopathic cholelithiasis. Niyogi et al.[9] showed an increasing incidence of children presenting with primary cholelithiasis in recent years in their two-surgeon, single-center study, with a median age of 13 years, a higher number of females, and most of the patients undergoing cholecystectomy laparoscopically. Nachulewicz et al.'s[10] study involved 149 three-port laparoscopic cholecystectomies at the specialist pediatric surgeon's practice, with no complications. St Peter et al.[11] performed laparoscopic cholecystectomy upon 224 children of higher age groups, with all having symptomatic relief after the procedure and a minimal complication rate. Novielloa et al.[12] showed familiarity and obesity to be the major predisposing factors nowadays for pediatric cholelithiasis. Kumar et al.[13] found no identifiable risk for cholelithiasis in most of the patients in their study group, while ceftriaxone usage was implicated in the minority; also, anemia was of concern among the children included. Bhasin et al.[14] treated higher age group pediatric patients in their study, with more of male patients; the cause was idiopathic in most cases and most of the gallstones were of mixed type. Majority of the patients in Jogendra's[15] study did not have any identifiable risk factors for cholelithiasis. Maharjan et al.[16] found no identifiable cause for cholelithiasis among pediatric patients undergoing cholecystectomy by general surgeons, with a minimal complication rate. Carbajo et al.[17] described the common congenital biliary anomalies likely to be encountered during cholecystectomies and advised adequate caution during surgery to avoid complications.

The age of patients in our study, presenting at the general surgeon's hospital (KA), was obviously higher, among the “pediatric” age group, along with a higher number of adult patients, as opposed to a specialist pediatric surgeon's practice (AP), catering to lower age group patients (agemedian AP: KA = 9:11 years) [Table 1]. Congenital anomalies of the biliary system may sometimes predispose to the formation of cholelithiasis in the pediatric age group itself, as found in one of the patients having a “double” (bilobed) gallbladder, with both the “bodies” of the complex filled up with variously sized calculi [Figure 1].[1],[17] “Phrygian cap” variant was found among few of the cases, which had no pathological significance.
Figure 1: A “double” (bilobed) gallbladder, part of a congenital duplication anomaly, in a 14-year-old female having undergone open cholecystectomy (anterograde approach) for calculous cholecystitis (KA). Ultrasonography abdomen was suggestive of multiple calculi inside an abnormally large-sized (for age) gallbladder, with pericholecystic adhesions. (a) Two separate bodies and fundus of the gallbladder, fused toward the neck part, with a common short cystic duct. (b) The calculi (mixed type) was retrieved after opening up the gallbladder bodies, post excision; the larger set from part 1 and the smaller set of stones from part 2 of the gallbladder complex

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  Conclusion Top


Symptomatic pediatric gallstone disease is increasingly being diagnosed as a cause of pain abdomen in children. Changing epidemiologic trends in recent years show that most cases are idiopathic or obesity/diet related in today's clinical practice, and occur at a younger age. Operative intervention under an experienced surgeon, to prevent morbidity and complications, is a safe treatment strategy, translating to a favorable change in public perception. Specialist pediatric surgeons are better equipped and trained to manage play-age-group patients, whereas older children can be managed by general surgeons.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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Emedicine Medscape. Pediatric Gallbladder Disease Surgery. Available from: https://emedicine.medscape.com/article/936591-overview, Pediatric Gallstones (Cholelithiasis). Available from: https://emedicine.medscape.com/article/927522-overview, Gallbladder Anatomy. Available from: https://emedicine.medscape.com/article/1900182-overview#showall. [Last accessed on 2020 May 31].  Back to cited text no. 1
    
2.
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Holcomb GW 3rd, Morgan WM 3rd, Neblett WW 3rd, Pietsch JB, O'Neill JA Jr., Shyr Yu. Laparoscopic cholecystectomy in children: Lessons learned from the first 100 patients. J Pediatr Surg 1999;34:1236-40.  Back to cited text no. 7
    
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Esposito C, Gonzalez Sabin MA, Corcione F, Sacco R, Esposito G, Settimi A. Results and complications of laparoscopic cholecystectomy in childhood. Surg Endosc 2001;15:890-2.  Back to cited text no. 8
    
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Niyogi A, Jeeneea R, Mehta N, Jones MO, Hey A. The changing face of paediatric cholecystectomy. Bulletin 2015;97:17-21. [doi: 10.1308/rcsbull. 2015.e17].  Back to cited text no. 9
    
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Nachulewicz P, Kasza A, Osemlak P, Wo źniak M, Pac-Kożuchowska E, Cielecki C, et al. Laparoscopic cholecystectomy in children: One centre experience. Elsevier science direct. Pediatr Pol 2016;91:118-21. [doi: 10.1016/j.pepo.2015.12.004].  Back to cited text no. 10
    
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St Peter SD, Keckler S, Nair A, Andrews WS, Sharp RJ, Snyder CL, et al. Laparoscopic cholecystectomy in the pediatric population. J Laparoendosc Adv Surg Tech A 2008;18:127-30.  Back to cited text no. 11
    
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Carbajo MA, Martín del Omo JC, Blanco JI, Cuesta C, Martín F, Toledano M, et al. Congenital malformations of the gallbladder and cystic duct diagnosed by laparoscopy: High surgical risk. JSLS 1999;3:319-21.  Back to cited text no. 17
    


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