Objective: To report a case of true agenesis of the gallbladder (GB) with obstructive jaundice due to primary choledochal stone. Clinical Presentation and Intervention: A 66-year-old woman presented with a full picture of obstructive jaundice, both clinically and biochemically. Ultrasound and endoscopic retrograde cholangiopancreatography failed to show the GB but showed dilatation of extrahepatic ducts with a stone in the common bile duct (CBD). Upon exploration, the GB was found to be absent and through a choledochotomy, the stone in the CBD was removed. Conclusion: This case shows that proper exploratory laparotomy and intraoperative cholangiography may be required to confirm absence of the GB.
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