Primary choledocholithiasis after cholecystectomy without hyperbilirubinaemia in a child with sickle cell disease

Ashley Inez Thrower1, Annette Roberts2, Joshua P Lee3

  • 1Internal Medicine and Pediatrics, Duke University, Durham, North Carolina, USA.

BMJ Case Reports
|April 16, 2025
PubMed

Insights

This study highlights a rare case of primary choledocholithiasis in a child with sickle cell disease (SCD) post-cholecystectomy. Normal bilirubin levels can mask this serious biliary tract condition in SCD patients.

Area of Science:

  • Pediatric Gastroenterology
  • Hematology
  • Hepatobiliary Medicine

Background:

  • Children with sickle cell disease (SCD) are prone to hepatobiliary issues due to chronic hemolysis and sickling.
  • Choledocholithiasis in pediatric SCD typically arises from gallstones, but primary stones can occur.
  • Prior cholecystectomy and normal bilirubin levels can impede diagnosis of biliary disease in SCD.

Purpose of the Study:

  • To report an uncommon instance of primary choledocholithiasis in a pediatric patient with SCD.
  • To emphasize the diagnostic challenges posed by normal bilirubin levels and a history of cholecystectomy.

Main Methods:

  • Case report detailing clinical presentation, diagnostic workup, and management.
  • Review of relevant literature on biliary tract pathology in pediatric sickle cell disease.

Main Results:

  • A child with SCD and a history of cholecystectomy presented with abdominal pain and primary choledocholithiasis.
  • Diagnostic delays were noted due to normal conjugated bilirubin levels.

Conclusions:

  • Primary choledocholithiasis is a rare but possible diagnosis in children with SCD, even after cholecystectomy.
  • Healthcare providers must maintain a high index of suspicion for biliary disease in SCD patients with abdominal pain, irrespective of bilirubin levels or prior surgery.

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