Objective: Rare diseaseBackground: Endogenous bacterial endophthalmitis is caused by a breach of the blood-ocular barrier by pathogens originat-ing from distant infective foci. Here, we report a case of endogenous endophthalmitis due to cholangitis com-plicated by common bile duct stones, which is a rare source of infection.Case Report: A 73-year-old man with type II diabetes mellitus underwent endoscopic choledocholithotripsy 20 years ago and laparoscopic cholecystectomy 18 years ago. He had choledocholith-related cholangitis 6, 5, and 1 years previ-ously and 4 times in the last year and underwent endoscopic choledocholithotripsy each time. Three days af-ter the last surgery, the patient developed right endogenous endophthalmitis and vitrectomy was performed. Four months later, the patient relapsed with cholangitis and required surgery for recurrent endophthalmitis. Roux-en-Y choledochojejunostomy was performed with curative intent, and the patient was followed up for 5 years without recurrence of choledocholith, cholangitis, or endophthalmitis Conclusions: The recommended treatment strategy for patients diagnosed with common bile duct stones or choledocho-lithiasis is stone extraction. Endoscopic retrograde cholangiopancreatography (ERCP) with endoscopic inter-vention is a widely accepted procedure. However, in cases of recurrent choledocholithiasis, the rate of recur-rence increases and the interval between ERCP becomes shorter in proportion to the number of recurrences. In such intractable cases requiring numerous sessions of endoscopic stone removal, bypass Roux-en-Y choled-ochojejunostomy should be performed to prevent possible rare complications such as endogenous bacterial endophthalmitis.