Case Report on Isolated Primary Biliary Tuberculosis: Rare Etiology of Indeterminate Biliary Stricture

Tuberculosis infection of the biliary tract is extremely rare. Pathogenesis of extra hepatic biliary tuberculosis includes extension from affected structures and rarely hematogenous, like infection from the portal tracts of liver is the most common route of spread, followed by infection from tuberculosis of periportal lymph nodes and ascending infection through the ampulla of Vater. In our case a 60-year-old man from South India presented to our hospital with complaints of jaundice for 2 months, upper abdominal pain for 10 days associated with pale-colored stools and itching. He had history of global loss of appetite and loss of weight 8 kg in 2 months. There was no history of surgery or other comorbidities. Past history was unremarkable. No history of native medication intake and blood transfusion or sexual promiscuity. General examination revealed jaundice and scratch marks all over the body. Abdomen examination was normal. Routine base line blood investigations suggest; Hb 10.5, TLC 6500, PLT 1.98000. ESR was 90 mm/h. Liver function test suggests conjugated hyper bilirubinemia with deranged enzymes and altered albumin and globulin ratio. Viral markers for hepatitis, HIV -I and II were negative. Normal renal function test and serum electrolytes. Chest X- ray was normal. USG whole abdomen showed normal CBD and CHD but dilated IHBR and small poorly demarcated hypoechoic lesion in right hepatic lobe. No evidence of chronic liver disease. CECT abdomen with triphasic CT showed stricture in right hepatic duct with cholangitis and cholangitic microabcess of 9 mm to 10 mm in right lobe of liver-possibility of malignant stricture (periductal infiltrating type of cholangiocarcinoma with cholangitis also considered). Ca 19-9 was normal (18.6 U/mL). Serum IgG4 was normal (0.08 gm/L). Serum ANA, ANCA-MPO were negative (1.5 RU/mL). ERCP was performed. Cholangiogram showed smooth narrowing and abnormal mucosa at right hepatic duct and dilated proximal segment and dilated intrahepatic biliary radicles in right lobe. CBD and CHD and left hepatic duct were not dilated. With the help of cholangioscope entire CBD, CHD were visualized. There was nodularity, sclerosis, fluffy overhanging tissue seen in right hepatic duct 1 cm from bifurcation. Hepatic duct orifices were normal. Using Spyglass DS II targeted biopsies were taken from right hepatic duct narrowing by spy bite forcep. There was resistance while negotiating biliary stent across the stricture. After that dilatation of right hepatic duct stricture was attempted by biliary dilators and metal Sohendra dilator. Post-dilatation two double pigtail plastic biliary stents were deployed into right hepatic duct. Post procedure free bile flow noted and no immediate postprocedure complication seen. Histopathology report suggested of benign bile duct epithelium and cluster epitheloid granulomata with necrosis as well as Langerhans giant cell. No evidence of dysplasia or malignancy. Patient was started anti tubercular regimen for six months. After starting treatment dramatic improvement of physical well-being and quality of life noted. Repeat liver function test showed improvement of bilirubin and liver enzymes. Currently patient is on regular follow up. Biliary tuberculosis should be considered as differential diagnosis, especially in a high endemic country with indeterminate biliary stricture and atypical presentations.

Publication History

Article published online:
22 April 2024