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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 3  |  Issue : 2  |  Page : 150-153

Primary biliary tuberculosis: A case report and review of literature


1 Department of Surgical Gastroenterology, Narayana Medical College, Nellore, Andhra Pradesh, India
2 Department of Pathology, Narayana Medical College, Nellore, Andhra Pradesh, India

Date of Web Publication13-Dec-2013

Correspondence Address:
S M Sivaraj
Department of Surgical Gastroenterology, Narayana Medical College, Nellore - 524 002, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-9596.122943

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  Abstract 

Primary biliary tuberculosis in the absence of liver involvement is a rare form of hepatobiliary tuberculosis. Barely 20 cases were reported in world literature. We present a 21-year-old man who presented to our hospital with upper abdominal pain and features of obstructive jaundice of 2 months duration. Following clinical evaluation, his radiological imaging revealed common bile duct stricture with proximal dilatation. Clinical diagnosis of cholangiocarcinoma was made and the patient underwent resection and hepaticojejunostomy. Histological examination revealed biliary tuberculosis. The patient was placed on anti-tuberculosis drugs. After one year of follow-up, patient is symptom free. Even though biliary tuberculosis is a rare cause of obstructive jaundice, in endemic areas, it should be considered as a differential diagnosis and investigated, as it can be potentially curable.

Keywords: Biliary, extra pulmonary tuberculosis, tuberculosis


How to cite this article:
Sivaraj S M, Sivacharan P, Mallikarjuna G R, Vamsikrishna P R, Ramakrishna B A, Thirunavukkarasu S. Primary biliary tuberculosis: A case report and review of literature. Arch Int Surg 2013;3:150-3

How to cite this URL:
Sivaraj S M, Sivacharan P, Mallikarjuna G R, Vamsikrishna P R, Ramakrishna B A, Thirunavukkarasu S. Primary biliary tuberculosis: A case report and review of literature. Arch Int Surg [serial online] 2013 [cited 2024 Mar 29];3:150-3. Available from: https://www.archintsurg.org/text.asp?2013/3/2/150/122943


  Introduction Top


Gastrointestinal tuberculosis is still a common diagnosis in clinical practice in developing countries. Common sites involved are ileo-cecal region, peritoneum, and mesenteric lymph nodes. Hepatobiliary tuberculosis is one of the uncommon presentations of abdominal tuberculosis and isolated biliary tuberculosis is the rarest form of hepatobiliary tuberculosis. [1] We present a rare case of extra hepatic biliary tuberculosis, which presented with features of obstructive jaundice.


  Case Report Top


A 21-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-coloured stools and itching. He had lost weight. There was no history of surgery or other comorbidities. General examination revealed jaundice and scratch marks all over the body. Abdominal examination revealed a palpable gall bladder. The liver was enlarged about 6 cm but there was no ascites. Digital rectal examination was normal except for pale-coloured stool. A clinical diagnosis of malignant obstructive jaundice was made. He had a total bilirubin of 12 mg/dl (Normal range-0.2-1mg/dl), the direct component was 9.8 mg/dl (Normal range-0-0.4 mg/dl), and alkaline phosphatase was elevated to 1003 U/L (Normal range-64-306 U/L). Screening for HIV and HBV were negative. Ultrasound examination of the abdomen revealed mild hepatomegaly with distended gall bladder and a dilated common bile duct of 1.7 cm at porta hepatis. Magnetic resonance cholangio pancreatography showed abrupt cutoff below cystic duct-hepatic junction and distended gall bladder with dilated common bile duct and intra hepatic biliary ducts. Magnetic Resonance Imaging (MRI) showed enlarged multiple pericholedochal and common hepatic lymphnodes. Serum Carcino-embryonic antigen was 12 ng/ml (Normal <5ng/ml) and CA 19.9 was 86 U/ml (Normal <37 U/ml).

With the preoperative diagnosis of cholangiocarcinoma, exploratory laparotomy was carried out, which revealed a biliary stricture beginning from cystic duct-CHD junction extending 3 cm down the CBD [Figure 1] with multiple lymph nodes; hepatoduodenal nodes and common hepatic nodes measuring 12 mm and firm in consistency. There were no liver metastases or peritoneal seedlings. Resection of the tumor with lymphadenectomy, frozen section of distal margin, and closure of distal end with hepaticojejunostomy of proximal end was done.
Figure 1: Operative photograph of biliary stricture

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Histopathological examination of the resected tumor showed features of thickened ulcerated CBD mucosa [Figure 2] with underlying stromal tissue with plenty of epitheloid granulomas [Figure 3], Langhans giant cells, fibroblasts, lymphocytes, plasma cells with central necrosis suggestive of tuberculosis of the common bile duct. Sections from lymph nodes showed plenty of epitheloid cell granulomas with caseous necrosis [Figure 4].
Figure 2: Microphotograph showing ulcerated lining of common bile duct (H and E ×100)

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Figure 3: Micorphotographs showing epithelial glands and granulomas in the biliary duct (H and E ×100)

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Figure 4: Microphotograph of lymphnode showing multiple granuloma with caseating necrosis (H and E ×100)

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Clinical examination and investigations like MRI of abdomen and chest xray did not reveal any other organ involvement with tuberculosis. Patient was started on antituberculosis regimen (Isoniazid, rifampicin, ethambutol and pyrazinamide) for six months, resulting in weight gain and symptom free after one year of followup.


  Discussion Top


Tuberculosis of the liver is quite common, but isolated tuberculosis of the extra hepatic biliary tree is rare. Hepatic tuberculosis was reported by Stemmerman in 1945. [2] Hepatic tuberculosis is reported to occur in 50-80% of patients, who had pulmonary tuberculosis related mortality. [3] In an autopsy study of 150 adult cases of tuberculosis over a period of 3 years, 42% of the patients showed epitheliod granulomas in the liver. [4] However, the first case of extra hepatic biliary stricture due to tuberculosis involvement of bile duct was reported by Fan et al., only in 1989. [5]

Pathogenesis

Pathogenesis of extra hepatic biliary tuberculosis includes extension from affected structures and rarely haematogenous. Descending 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. [6] Our case, had no other organ involvement that could be diagnosed by MRI of the abdomen and chest xray. Other reports indicated 28% to 64% cases with other organ involvement. [1],[7]

Presentation and evaluation

Tuberculous biliary stricture can be an isolated stricture of extra hepatic biliary tree, as presented in our case or may be a more common complex multiple strictures involving the hilar region. Biliary involvement due to granulomatous hepatitis are usually associated with multiple and complex strictures. [7] Associated malignancy with tuberculous stricture has also been reported. [8] The symptoms of tuberculosis can result from primary tuberculosis of bile duct causing stricture, ruptured caseating granuloma into the bile duct, compression of bile duct by tuberculous adenitis, postinflammatory stricture or from compression by tuberculous psuedotumor. [9] Clinical presentations include chronic abdominal pain, obstructive jaundice, itching, fever, vomiting, anorexia, weight loss and hematemesis. [10]

Imaging studies do not provide definitive diagnosis of tuberculosis. [11] In our case also preoperative diagnosis of tuberculosis was not made. Most cases are reported following postoperative histopathological examination. Preoperative diagnosis is by high index of suspicion in view of atypical presentation or tuberculosis involvement of other organs. Imaging by US, CT, MRCP are useful in identifying the level and extent of bile duct involvement, and may identify any inflammatory mass or lymphadenitis. [11] Endoscopic retrograde cholangiopancreatography (ERCP) or Percutaneous transhepatic cholangiogram features of biliary TB are pruning of distal intrahepatic duct, hilar strictures with dilated intrahepatic biliary radicles, long smooth stricture of the distal biliary duct and sclerosing cholangitis like changes. [6] However, they are seldom pathognomonic of the disease. Only a positive tissue sample can give a definitive preoperative diagnosis. Tissue diagnosis can be obtained by ERCP and bile aspirate microscopy, US or CT guided percutaneous biopsy or choledochoscopic biopsy through percutaneous biliary canulation of the dilated ducts. In view of short duration of presentation the most common aetiology, malignant stricture was considered and percutaneous biopsy was not done. Acid Fast Bacteria (AFB) staining of bile aspirate has low yield (20%) and only few studies are reported. [12],[13],[14] Polymerase chain reaction (PCR) for percutaneous liver biopsy of liver tuberculosis had 88% overall positivity, but there are few reports only on PCR of ERCP aspirate, for biliary tuberculosis. [13],[14] In view of low resources, those investigations were not done.

Treatment

In established cases ATT will cure tuberculosis in nearly all cases. But in most cases biliary tuberculosis may heal with stricture. [12],[15] Patients may require endoscopic stenting or surgery (bilioenteric anastomosis) for management of the stricture. As was the case in our patient, operation is also required if preoperatively the surgeon is unable to rule out malignancy. Extensive strictures with intrahepatic extension may not be amenable to surgery requiring definitive endoscopic stenting by metallic stents. [13] Recent case series reported satisfactory results following surgical management of these inflammatory strictures which were initially being managed by endoscopy. [11],[15]

Biliary tuberculosis should be suspected in young patients who do not fall in the age group of malignant obstructive diseases, had other organs with tuberculosis involvement and in high endemic areas. Curable diseases like biliary tuberculosis should be considered as differential diagnosis, especially in a high endemic country with atypical presentation. These cases may benefit from preoperative tissue biopsy or bile aspirate polymerase chain reaction that can be followed by antituberculous therapy.

 
  References Top

1.Chong VH. Hepatobiliary tuberculosis: A review of presentations and outcomes. South Med J 2008;101:356-61.  Back to cited text no. 1
[PUBMED]    
2.Stemmerman M. Bile duct tuberculosis. Q Bull Sea View Hosp 1941;6:316-24.  Back to cited text no. 2
    
3.Hussain W, Mutimer D, Harrison R, Hubscher S, Neuberger J. Fulminant hepatic failure by tuberculosis. Gut 1995;36:792-4.  Back to cited text no. 3
[PUBMED]    
4.Amarapurkar A, Agrawal V. Liver involvement in tuberculosis-an autopsy study. Trop Gastroenterol 2006;27:69-74.  Back to cited text no. 4
[PUBMED]    
5.Fan ST, Ng IO, Choi TK, Lai EC. Tuberculosis of bile duct: A rare cause of biliary stricture. Am J Gastroenterol 1989;84:413-4.  Back to cited text no. 5
[PUBMED]    
6.Alvarez SZ, Carpio R. Hepatobiliary tuberculosis. Dig Dis Sci 1983;28:193-200.  Back to cited text no. 6
[PUBMED]    
7.Chong VH, Lim KS. Hepatobiliary tuberculosis. Singapore Med J 2010;51:744-51.  Back to cited text no. 7
[PUBMED]    
8.Chong VH, Telisinghe PU, Yapp SK, Jalihal A. Biliary stricture secondary to tuberculosis and early ampullary carcinoma. Singapore Med J 2009;50:e94-6.  Back to cited text no. 8
[PUBMED]    
9.Amarapurkar DN, Patel ND, Amarapurkar AD. Hepatobiliary tuberculosis in western India. Indian J Pathol Microbiol 2008;51:175-81.  Back to cited text no. 9
[PUBMED]  Medknow Journal  
10.Govindasamy M, Srinivasan T, Varma V, Mehta N, Yadav A, Kumaran V, et al. Biliary tract tuberculosis-a diagnostic dilemma. J Gastrointest Surg 2011;15:2172-7.  Back to cited text no. 10
[PUBMED]    
11.Saluja SS, Ray S, Pal S, Kukeraja M, Srivastava DN, Sahni P, et al. Hepatobiliary and pancreatic tuberculosis: A two decade experience. BMC Surg 2007;7:10.  Back to cited text no. 11
[PUBMED]    
12.Bearer EA, Savides TJ, McCutchan JA. Endoscopic diagnosis and management of hepatobiliary tuberculosis. Am J Gastroenterol 1996;91:2602-4.  Back to cited text no. 12
[PUBMED]    
13.Alcantara-Payawal DE, Matsumara M, Shiratori Y, Okudaira T, Gonzalez R, Lopez RA, et al. Direct detection of mycobacterium tuberculosis using polymerase chain reaction assay among hepatic granuloma. J Hepatol 1997;27:620-7.  Back to cited text no. 13
    
14.Probst A, Schmidbaur W, Jechart G, Hammond A, Zentner J, Niculescu E, et al. Obstructive jaundice in AIDS: Diagnosis of biliary tuberculosis by ERCP. Gastrointest Endosc 2004;60:145-8.  Back to cited text no. 14
[PUBMED]    
15.Inal M, Aksungur E, Akgul E, Demirbas O, Oguz M, Erkocak E. Biliary tuberculosis mimicking cholangiocarcinoma: Treatment with metallic biliary endoprosthesis. Am J Gastroenterol 2000;95:1069-71.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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