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We report the case of a 16-year-old boy with primary sclerosing cholangitis associated with inflammatory bowel disease who was initially treated and controlled pharmacologically. He underwent living donor liver transplantation (LDLT) after he developed progressive biliary tract abnormalities and portal hypertension accompanied by recurrent bile duct infections. Two months following LDLT, the hepaticojejunostomy anastomosis became occluded and it was treated surgically. Few weeks later, an increase in drain production persisted without focus; therefore, further diagnostic tests were conducted which proved the recurrence of biliary cast. Under sonographic guidance, external drainage of bile was carried out by percutaneous transhepatic cholangiography and drainage. In total, our patient underwent 5 interventions under general anesthesia and clinically, our patient's general condition improved, and he gained weight. Minimally invasive procedures such as percutaneous transhepatic cholangiography and drainage and balloon dilation are safe and effective, but may require several attempts before being successful. Keywords: Biliary strictures, Pediatric liver transplantation, Percutaneous transhepatic cholangiography and drainage