Cystic Duct Perforation: A Potential Complication of Choledocholithiasis in Infants

Choledocholithiasis with cystic duct perforation is a rare event in infants, particularly those without underlying risk factors. A 5-month-old male, with no prior health issues, presented with poor weight gain, irritability, and abdominal pain. Investigations revealed leukocytosis and mild hyperbilirubinemia, while imaging showed intra-abdominal free fluid without gallstones. Laparoscopy illustrated bilious ascites, and laparotomy confirmed a distal common bile duct stone with cystic duct perforation.

The patient underwent open trans-cystic bile duct exploration, cholecystectomy, and cystic duct ligation, with intraoperative cholangiogram confirming biliary patency. He recovered uneventfully, tolerated oral feeds by day 4, and was discharged on day 11. Follow-up demonstrated weight gain and no recurrence. This case highlights cystic duct perforation as a rare but important complication of infantile choledocholithiasis, requiring timely surgical intervention for favorable outcomes.

• A 5-month-old infant (weight 5.9 kg) presented to the emergency care center with a 1-week history of persistent fussiness, progressive abdominal distension, and reduced stool output.
• His mother also reported episodes of intermittent vomiting prior to admission, which improved after switching to smaller-volume feeds.
• In addition, the mother noticed that the infant had passed pale, acholic (clay-colored) stools during this period.

Introduction

In the pediatric population, cholelithiasis (gallstone formation) and choledocholithiasis (presence of stones within the common bile duct) are uncommon. The overall prevalence of cholelithiasis in children is estimated at approximately 4%. Data on the frequency of choledocholithiasis are limited, although a few reports suggest that fewer than one-third of pediatric patients undergoing cholecystectomy present with choledocholithiasis.

In infants, gallstones are most often linked with underlying ailments such as hemolytic disorders, sepsis, congenital biliary malformations, inherited hyperbilirubinemia, prematurity, or exposure to certain medications. In many cases, gallstones in infants and children are incidentally detected in otherwise asymptomatic patients.

This case report describes the case of a 5-month-old infant without identifiable risk factors for gallstone disease, who developed cystic duct perforation secondary to an impacted gallstone in the distal common bile duct.

Medical History

The infant had a significant history of growth restriction, with weight at the 1st percentile and height below the 1st percentile.​​​​​​​​​​​​​​

• On initial evaluation, the infant appeared comfortable and in no acute distress, and his temperature and vital signs were within normal limits for age.
• His abdomen was visibly distended and was mildly tender to palpation, although there was no guarding or rebound tenderness to suggest peritonitis.
• His sclerae appeared anicteric, and there were no stigmata of chronic liver disease. Laboratory assessments depicted leukocytosis with a white blood cell count of 15.14 × 10³/mcL.
• Liver function tests illustrated a mildly elevated total bilirubin level of 1.7 mg/dL, alkaline phosphatase of 230 U/L, alanine aminotransferase of 75 U/L, and aspartate aminotransferase of 56 U/L, while the rest of the serum biochemical studies were within normal limits.
• A plain abdominal radiograph demonstrated non-obstructive gaseous distension of the bowel loops.
• Abdominal ultrasound revealed no features of obstruction or intussusception, but it did show a moderate amount of free intraperitoneal fluid with internal debris, raising suspicion for a possible perforated viscus.
• A computed tomography (CT) scan of the abdomen and pelvis confirmed the presence of moderate free fluid and illustrated non-opacified bowel loops in the mid-abdomen, with the additional possibility of a stone in the common bile duct that could not be excluded.

• Owing to concerns for bowel or biliary perforation, the infant was taken urgently to the operating room for a diagnostic laparoscopy.
• Upon entry, a large volume of bilious ascites (a condition where bile leaks into the abdominal cavity, causing fluid buildup or ascites) was encountered, and approximately 400 cc of bilious fluid was aspirated.
• The appendix and bowel were inspected and found to be grossly normal. However, bile staining and significant edema were noted in the right upper quadrant around the gallbladder and porta hepatis.
• These findings prompted conversion to an open laparotomy through a right subcostal incision for further exploration.
• Intraoperative cholangiography revealed a completely obstructing stone in the common bile duct, with extravasation of contrast near the cystic duct–common bile duct junction, confirming perforation at this site.
• Endoscopic retrograde cholangiopancreatography (ERCP) was not feasible given the infant’s small size and low weight. Hence, a trans-cystic common bile duct exploration was executed under fluoroscopic guidance.
• The obstructing stone was removed successfully using a 2Fr Fogarty catheter, and restoration of duct patency to the duodenum was confirmed with a repeat cholangiogram.
• The cystic duct was ligated proximal to the site of perforation, and a cholecystectomy was subsequently performed.
• The peritoneal cavity was irrigated with copious saline to clear the bilious contamination, and a 15Fr Blake drain was positioned adjacent to the common bile duct prior to abdominal closure.
• Pathologic examination of the resected gallbladder confirmed acute and chronic cholecystitis with numerous gallstones.
• In the postoperative period, the infant was given intravenous ceftriaxone for 4 days to address the risk of intra-abdominal infection due to bile spillage.
• He remained clinically stable, without development of fever or worsening leukocytosis.
• Given his ongoing failure to thrive, peripheral parenteral nutrition was given from postoperative day (POD) 2 to POD 4 for nutritional support.
• Oral intake was resumed with clear fluids on POD 4 and gradually advanced to breast milk and formula on POD 5.
• The Blake drain was removed on POD 5 once the infant showed tolerance to enteral feeding and bilious drainage had subsided.
• He remained hospitalized for a total of 11 days after surgery to allow close monitoring and nutritional optimization.
• At 1-year follow-up, the child remained free of postoperative complications.
• His growth parameters improved markedly, with his weight-for-age increasing to the 21st percentile and his length-for-age to the 33rd percentile.
• Follow-up right upper quadrant ultrasounds depicted no recurrent or residual biliary pathology.
• Extensive metabolic and hematologic evaluation to investigate the etiology of gallstone formation in this infant did not reveal any underlying abnormality.

Discussion

Cholelithiasis and choledocholithiasis are rare in infants, and gallstone formation without predisposing factors is extremely uncommon. Biliary perforation, most often connected with choledochal cysts in infants and children, has a reported prevalence of 1–7%. Reports of biliary perforation in infants are scarce, and to date, no cases describing the combination of choledocholithiasis, cystic duct perforation, and biliary peritonitis in a 5-month-old infant have been published.

Diagnosis in this age group is cumbersome since infants often lack the classic symptoms seen in adults. Only 15% of infants under 1 year with gallstones are symptomatic. Clinical suspicion is especially fundamental in infants with risk factors such as Trisomy 21 or hemolytic disease, though the reported prevalence of these risk factors ranges widely from 10% to 60%. Laboratory testing has limited reliability in infants. In 1 retrospective review, only aspartate aminotransferase and alkaline phosphatase levels were markedly different in kids with gallstones compared to controls, while bilirubin and white blood cell counts showed no distinction.

Many of these studies include older children and adolescents, which minimizes the applicability to infants. Management strategies remain inconsistent. Both non-surgical and surgical approaches are described. Non-surgical options include observation or treatment with ursodeoxycholic acid (UDCA). While some studies report up to 90% dissolution rates and advocate UDCA for asymptomatic patients, others suggest restricting its usage to symptomatic children who are not surgical candidates.

For obstructive common bile duct stones, management options encompass ERCP or surgical common bile duct exploration. ERCP is technically feasible in children, with 1 study showing 100% success and a 10% adverse event rate. However, data on infants are limited, and no standardized pediatric guidelines exist. Consequently, surgery is often required. In the presented case, due to the infant’s small size, open trans-cystic fluoroscopic-guided common bile duct exploration was performed using a Fogarty catheter to clear the distal obstruction.

Learning

• Cystic duct perforation is a rare but serious complication of choledocholithiasis in infants.
• Infants may present with subtle, non-specific symptoms such as irritability, abdominal distension, poor feeding, or pale stools.
• Standard imaging may fail to identify stones, making surgical exploration often necessary for diagnosis.
• Timely operative intervention, including trans-cystic common bile duct exploration and cholecystectomy, can result in full recovery and improved growth.
• Clinicians should maintain high suspicion for biliary pathology even in infants without known risk factors.