Percutaneous transhepatic biliary drainage (PTBD) has been widely used, but it has a potential risk of tumor spread along the catheter tract. We herein present a case of solitary PTBD tract metastasis after curative resection of perihilar cholangiocarcinoma. Initially, endoscopic nasobiliary drainage was done on a 65 year-old female patient, but the cholangitis did not resolve. Thus a PTBD catheter was inserted into the right posterior duct. Right portal vein embolization was also performed. Curative surgery including right hepatectomy and bile duct resection was performed 16 days after PTBD. After 12 months, serum CA19-9 had increased gradually without any symptoms. Finally, a small right pleural metastasis was found through strict tumor surveillance for 6 months. Chemoradiation therapy was performed, but there was no response to treatment. As the tumor progressed, she complained of severe dyspnea and finally died from tumor dissemination to the chest and bones 18 months after the first detection of PTBD tract recurrence and 36 months after surgery. No intra-abdominal recurrence was found until the terminal stage. This PTBD tract recurrence was attributed to the PTBD even though it was in place for only 16 days. Although such recurrence is rare, its risk should be taken into account during follow-up of patients who have received PTBD before.