Oriental cholangiohepatitis (OCH) is a disease characterized by intrabiliary pigment stone formation, resulting in recurrent bouts of cholangitis. OCH is found mostly in Southeast Asia but it is occasionally recognized in Western societies. OCH etiology is largely unknown. We report our experience with a patient who presented with acute cholecystitis. Following laparoscopic cholecystectomy, she developed acute cholangitis due to multiple biliary tree stones. She underwent ERCP to clear the stones from common bile duct. For the intrahepatic stones, she underwent novel hybrid percutaneous endoscopic technique. The procedure resulted in complete clearance of biliary tree stones and resolution of her symptoms. The aim of this case is to increase awareness of this disease when patients from endemic areas present with biliary stones.
Oriental cholangiohepatitis (OCH) also called recurrent pyogenic cholangitis is a disease characterized by intrabiliary pigmented stones formation, resulting in biliary tree stricture and obstruction with recurrent bouts of cholangitis. OCH is found mostly in Southeast Asia (hence its name) but now occasionally recognized in Western societies with prevalence of less than 1% [
A 39-year-old Korean female presented with abdominal pain for 5 days. She was found to have acute cholecystitis after gallbladder ultrasound demonstrated cholelithiasis with wall inflammation. She underwent laparoscopic cholecystectomy with intraoperative cholangiogram which demonstrated choledocholithiasis. The following day, the patient developed fever and hypotension with elevated bilirubin level of 3.3 mg/dL. Her alkaline phosphatase was 174 U/L, alanine aminotransferase 215 U/L, and aspartate aminotransferase 126 U/L. She was started on IV antibiotics and had an urgent endoscopic retrograde cholangiopancreatography (ERCP) which demonstrated a common bile duct (CBD) stones with multiple intrahepatic stones as well (Figure
ERCP showing multiple CBD stones. Note that the right hepatic duct is not visualized due to obstruction.
Percutaneous cholangiogram showing multiple stones in biliary tree.
CT scan showing calcified stones within dilated intrahepatic ducts in the left lobe of the liver.
Cholangioscope image showing intrahepatic stone.
Percutaneous cholangiogram showing biliary tree after the procedure.
OCH is characterized by intractable nature and frequent recurrence requiring multiple operative interventions. In addition to frequent cholangitis and chronic sepsis, it is widely known that longstanding intrahepatic stones lead to intrahepatic cholangiocarcinoma which occurs in 5% of the cases [
OCH usually affects the left hepatic duct, especially the left lateral segmental duct, in the early course of the disease for unknown reason [
In conclusion, percutaneous endoscopic approach is relatively safe alternative therapy for intrahepatic stones extraction. Long-term follow-up is required, because the overall recurrence rate for intrahepatic stones and/or cholangitis is high (63.2%) especially in patients with bile duct stricture. Also, complete clearance of intrahepatic stones is crucial as the incidence of cholangiocarcinoma is significantly higher in those with residual stones [
Informed consent was obtained prior to publication of this case.
Muslim Atiq is the guarantor of this article. Khalil Aloreidi is the first author.
The authors have no conflicts of interest to declare.
Khalil Aloreidi is responsible for drafting and final approval of the manuscript. Prince Sethi is responsible for drafting and revisions and final approval of the manuscript. Terry Yeager contributed to concept and final approval of the manuscript. Muslim Atiq contributed to concept, drafting, and revisions and final approval of the manuscript.