Abstract

INTRODUCTION: Recurrent pyogenic cholangitis (RPC) is a disease predominantly found in Asian patients. It is the result of biliary stasis and stone formation in the proximal biliary tree due to strictures resulting in intrahepatic ductal dilatation, contrary to typical western biliary obstruction presentation. CASE DESCRIPTION/METHODS: A 79-year-old lady, immigrant from China, presented to the emergency department with altered mentation, jaundice, and abdominal pain. Patient has a pertinent history of cirrhosis, chronic hepatitis B, and numerous prior admissions for choledocholithiasis and ascending cholangitis over a period of eight years requiring multiple endoscopic retrograde cholangio-pancreatography (ERCP) procedures and common bile duct (CBD) stenting. On presentation, she was febrile, hypotensive, and tachycardic with right upper quadrant tenderness. Labs demonstrated leukopenia, lactic acidosis, and direct bilirubinemia. CT scan of the abdomen (Figure 1) showed diffuse intrahepatic and extrahepatic biliary dilatation, which has progressed from prior imaging one year ago, and presence of two CBD stents inserted 2 months ago. The clinical picture was concerning for ascending cholangitis secondary to choledocholithiasis and stent malfunction. She was bolused with IV fluids, started on broad-spectrum antibiotics, and transferred to the intensive care unit. She was then taken for ERCP (Figure 2). Two stents were removed which was followed by passage of multiple stones of different sizes (Figure 3). The remainder of stones were removed with balloon sweep and a new stent was placed in CBD. Her presentation with numerous recurrent episodes of ascending cholangitis, biliary dilatation, and recurrent stone formation leads to the diagnosis of RPC. Patient was offered definitive treatment with interval cholecystectomy and choledochojejunostomy but she elected for non-surgical management. She continued to improve on antibiotic therapy and was eventually discharged home. DISCUSSION: RPC is a rare cause of ascending cholangitis in the US, typically seen in immigrants from Southeast Asia. The resulting stones are numerous and mainly composed of calcium bilirubinate. Patients get frequent exacerbations throughout their lives. Cirrhosis and portal hypertension can develop in severe cases. Acute management involves removal of the stones and management of secondary infections. Long term treatment involves regular surveillance or surgical hepatobiliary resection of affected area with biliary-enteric anastomosis.Figure 1.: CT abdomen with IV contrast showing (A): intrahepatic biliary dilatation, (B): distended gallbladder with cholelithiasis (asterisk) and CBD dilatation (arrow) up to 2.2 cm in diameter with two stents, (C): a coronal view of the hepatobiliary system and two stents extending from the proximal CBD to the duodenum.Figure 2.: ERCP fluoroscopy showing (A): significant extrahepatic biliary dilatation with filling defects (arrow) likely gallstones, (B): moderate intrahepatic biliary dilatation.Figure 3.: Endoscopic images from ERCP procedure showing (A): two CBD pigtail stents removal and exudative biliary drainage from the ampulla of Vater, (B): confluence of gallstones that passed into the duodenum after the extraction of the CBD stents.

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