Granulomatous cholangitis mimicking hilar cholangiocarcinoma: a case report
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CASE REPORT
Granulomatous cholangitis mimicking hilar cholangiocarcinoma: a case report Shigeru Fujisaki1,2* , Motoi Takashina1, Ken‑ichi Sakurai3, Ryouichi Tomita3 and Tadatoshi Takayama2
Abstract Background: Hilar biliary stricture caused by isolated fungal infections in immunocompetent patients are consid‑ ered to be extremely rare and difficult to the diagnose from the outset. Case presentation: We report a unique case of granulomatous cholangitis based on isolated biliary fungal infec‑ tion manifesting as obstructive jaundice and mimicking hilar cholangiocarcinoma in an immunocompetent woman. A 67-year-old Japanese woman was referred to our hospital for obstructive jaundice. She had been followed up for hypochondroplasia by the referring physician. Her total bilirubin level was 5.4 mg/dL. Viral hepatitis screening was found to be negative, and serum IgG4 was within normal limits; however, her CA19-9 level was high. Abdominal com‑ puted tomography revealed dilatation of the intrahepatic bile ducts. Abdominal echogram detected a solid mass in the hilar bile duct. Her magnetic resonance cholangiopancreatography has also revealed an abrupt stenosis of the pri‑ mary biliary confluence with upstream dilatation of the intrahepatic bile ducts. Endoscopic nasobiliary drainage was then performed to improve the obstructive jaundice. Although biliary cytology did not reveal malignant findings, the bile duct in the hilum showed severe stenosis, and hilar cholangiocarcinoma could not be completely excluded. The patient had a developmental disorder based on chondrodystrophy. To avoid excessive surgical stress, such as hepatic lobectomy, we performed resection of the extrahepatic bile duct and Roux-en-Y hepaticojejunostomy reconstruction. Intraoperative frozen sections of the resection margins were determined to be negative for tumor. The resected speci‑ men showed multiple strictures inside the common bile duct, numerous calculi in the lumen, and little free space. The final pathological diagnosis was granulomatous cholangitis due to fungal infection. The patient’s postoperative course was deemed uneventful. She was discharged from our hospital 23 days after surgery without antifungal treatment. Conclusions: For a unique case of granulomatous cholangitis based on isolated biliary fungal infection mim‑ icking hilar cholangiocarcinoma, we were able to avoid excessive invasion and performed appropriate surgical management. Keywords: Granulomatous cholangitis, Hilar cholangiocarcinoma, Fungal infection Background Biliary fungal infection has rarely been observed, even in susceptible patients with systemic candidiasis [1]. Moreover, there is little chance of systemic fungal infection in immunocompetent patients, and biliary fungal infection is unlikely.
We report a case in which we performed an operation for biliary stricture in the hilum region because hilar cholangiocarcinoma could not be ruled out. This was an extremely rare case of granulomatous cholangitis based on isolated biliary fungal infection as the
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