Isolated biliary tuberculosis: a rare case report and diagnostic challenges
Department of Internal Medicine, Texas Tech University Health Sciences Center El Paso, The Hospital of Providence-Transmountain, Texas, USA
Department of Internal Medicine, UHS Wilson Medical Center, New York, USA
Department of Internal Medicine, Services Institute of Medical Sciences, Lahore, Pakistan
Department of Internal Medicine, Vassar Brothers Medical Center, New York, USA
Department of Gastroenterology, UHS Wilson Medical Center, New York, USA
Department of Internal Medicine, Shalamar Medical and Dental College, Lahore, Pakistan
Abstract
Introduction and importance:
Biliary tuberculosis is a rare form of Mycobacterium tuberculosis infection, accounting for only 0.0–0.1% of all TB cases in certain settings. Its preoperative diagnosis is difficult due to nonspecific symptoms and the lack of specific imaging criteria. Often, it mimics other diseases like cancers and infections, complicating early detection.
Case Presentation:
A 51-year-old female with a past medical history of diabetes mellitus, hypothyroidism, and a biliary stricture following stent placement presented with nausea, vomiting, loss of appetite, and weight loss for 4 months. A CT scan revealed an indwelling common bile duct (CBD) stent, mild intrahepatic biliary ductal dilatation, and pneumobilia. An endoscopic retrograde cholangiopancreatography (ERCP) procedure was performed with stent exchange. Initial CBD biopsy showed chronic inflammation, but both biopsy and fine-needle aspiration (FNA) were negative for malignancy. A subsequent ERCP with additional biopsies also returned negative results for malignancy, though CBD brushing tested positive for M. tuberculosis.
Clinical discussion:
The diagnosis of biliary tuberculosis is challenging due to its nonspecific presentation. In this case, the positive result for M. tuberculosis in the CBD brushing led to the diagnosis, even after negative biopsy and FNA results. Early recognition of hepatobiliary tuberculosis is crucial as it enables conservative management with stents and anti-tuberculosis therapy (ATT).
Conclusion:
Biliary tuberculosis, although rare, should be considered in patients with unexplained biliary symptoms. Timely diagnosis through appropriate diagnostic procedures can lead to effective treatment with ATT and stenting, improving patient outcomes, and preventing more invasive treatments.
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Keywords: biliary tuberculosis, common bile duct, pneumobilia
Article notes
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Received 2025 Jan 13; Accepted 2025 Apr 21; Collection date 2025 Jul.
Boxed Text
HIGHLIGHTS
- Isolated biliary tuberculosis is a rare form of Mycobacterium tuberculosis infection, accounting for only 0.0–0.1% of all TB cases.
- The diagnosis of biliary tuberculosis is challenging due to its nonspecific presentation.
- Biliary brushing during ERCP plays a pivotal role in diagnosing biliary tuberculosis (BTB) if CBD biopsy and fine-needle aspiration (FNA) biopsy are negative.
- Early recognition of hepatobiliary tuberculosis is crucial as it enables conservative management with stents and anti-tuberculosis therapy (ATT).
Introduction
Mycobacterium tuberculosis is a contagious disease that primarily affects the lungs[1]. Among the various extrapulmonary manifestations of tuberculosis (TB), abdominal TB is quite common, involving the gastrointestinal tract, spleen, pancreas, hepatobiliary system, and abdominal lymph nodes[2]. Extrapulmonary TB can occur in isolation or alongside pulmonary TB, although it is rare for it to present as a localized condition[1]. Liver involvement in miliary TB is observed in approximately 50–80% of cases in patients who succumb to pulmonary tuberculosis[3]. However, the involvement of the biliary system is extremely rare[2]. Extrapulmonary tuberculosis is a significant contributor to the global tuberculosis burden, particularly in developing countries. Globally, it accounts for approximately 15–20% of all TB cases. Hepatobiliary TB (HBTB), a subset of abdominal TB, remains exceedingly rare but poses diagnostic challenges due to its nonspecific clinical. HBTB presents with symptoms that mimic other diseases such as infections and cancers, often leading to delayed diagnosis and treatment[4]. Diagnosis is challenging and frequently occurs postoperatively, based on histological findings of Langerhans giant cells, caseation necrosis, and the formation of epithelioid granulomas[5]. In some cases, TB can be diagnosed preoperatively through the identification of acid-fast bacilli (AFB) in biliary fluid aspirate obtained during ERCP[6]. Medical management is crucial for patients diagnosed preoperatively, while surgery is reserved for those with significant symptoms[7]. A high degree of clinical suspicion is necessary for a preoperative diagnosis[8]. Here, we present a case of isolated TB of the biliary tract in a 51-year-old female.
Case presentation
A 51-year-old female with a past medical history of diabetes mellitus, hypothyroidism, and biliary stricture status post (s/p) biliary stent placement presented with complaints of abdominal pain, nausea, and vomiting for the last 4 months. The pain was not associated with the food and was not relieved by any antacids or proton pump inhibitors. She also reported a low appetite and weight loss of 20 pounds in the last 4 months. Computed tomography (CT) scan of the abdomen with and without contrast showed an indwelling common bile duct (CBD) stent in position (Fig. 1). Concomitant mild intrahepatic biliary ductal dilatation was evident along with pneumobilia, presumably related to the indwelling stent. However, the intrahepatic and CBDs appear to demonstrate wall enhancement. Endoscopic retrograde cholangiopancreatography (ERCP) with stent exchange was performed (Fig. 2).
The bile duct was explored endoscopically. The sludge was swept from the duct. A CBD biopsy was performed that was negative for malignancy but showed evidence of chronic inflammation (Fig. 3).
The CBD brushings were also obtained during the ERCP; brushing showed chronic inflammation and was negative for any malignancy. Endoscopic ultrasound (EUS) was also performed that showed the small unilocular in the tail of the pancreas and enlarged peripancreatic lymph nodes, and the largest one was 15 mm. Fine-needle aspiration (FNA) was performed on cyst and lymph nodes and was negative for the malignancy.
The patient continued to lose weight and presented again in 4 months with similar symptoms. Cholangiogram showed the filling defect consistent with stone or sludge. ERCP was performed again and showed a single diffused biliary stricture in the lower third of the CBD; the structure was indeterminate. The biopsies were taken from the CBD, and the biliary stricture came again negative for malignancy (Fig. 4), but the CBD brushing was positive for mycobacterium tuberculosis (Fig. 5). The diagnosis of M. tuberculosis of the CBD was made as the patient does not have any other foci of TB infection given his clear chest X-ray, and gastric and colon biopsies were negative for AFB. The patient was referred to the infectious disease department for further management.(Table 1)
| Timeline | Modality | Finding | Result |
|---|---|---|---|
| 0 Month | CT abdomen | Indwelling CBD stent, Mild ductal dilation, Pneumobilia | Nonspecific |
| 1 Month | ERCP (initial) | Sludge clearance, stent exchange | No malignancy |
| 1 Month | CBD biopsy | Lymphoplasmacytic inflammation | Negative for malignancy |
| 1 Month | CBD brushing | Chronic inflammation | Negative for malignancy |
| 1 Month | EUS + FNA | Cyst in pancreas tail, enlarged LNs (15 mm), FNA) | Negative for malignancy |
| 4 Month | ERCP (repeat) | Diffuse stricture in lower CBD | Indeterminate |
| 4 Month | CBD biopsy | Inflammation | Negative for malignancy |
| 4 Month | CBD brushing | AFB-positive | Positive for M. tuberculosis |
| 4 Month | Chest X-ray, gastric/colonic biopsies | No signs of pulmonary or GI TB | Negative for TB |
| Post Diagnosis | Referred to infectious disease |
Discussion
Biliary tuberculosis (BTB) is an uncommon extrapulmonary manifestation of tuberculosis[8]. It can develop through three distinct pathways: (i) descending infection from portal tracts into the bile ducts, (ii) periportal lymphadenitis due to TB, and (iii) ascending infection from the Vater ampulla[9]. The symptoms of biliary tuberculosis often result from strictures caused by primary tuberculosis of the biliary tract, post- ATT effects, rupture of caseating granulomas into the bile ducts, compression from tuberculous adenitis associated with periportal, pericholedochal, or peripancreatic lymph nodes, or from a pseudotumor[9]. Previous reports of biliary stricture due to bile duct tuberculosis are extremely rare[10]. In comparison with previously published cases, our patient similarly presented with vague abdominal symptoms and recurrent strictures that mimicked malignancy. However, unlike some reports where diagnosis was established through lymph node FNA or earlier tissue sampling, this case required multiple interventions and ultimately relied on positive CBD brushing to reach a diagnosis, making it one of the more diagnostically delayed presentations in literature. The diagnosis of biliary TB can become challenging due to its nonspecific symptoms as in our case, the patient’s recurrent biliary strictures and non-specific symptoms such as abdominal pain, weight loss, and nausea, along with negative malignancy results from multiple biopsies, posed a significant diagnostic challenge. An important limitation in this case was the initial failure of both lymph node FNA and multiple biopsy samples to detect TB, likely due to the patchy or localized involvement of the disease. These false-negative results significantly delayed appropriate treatment and underscore the low sensitivity of conventional histopathology and cytology in detecting M. tuberculosis in biliary tissues. The difficulty in diagnosing BTB can further increase by the overlapping clinical and radiological features with other biliary pathologies, such as cholangiocarcinoma and primary sclerosing cholangitis when hepatic granuoloma becomes involved leading to nuemerous complex strictures[11].
Preoperative diagnosis of BTB is crucial as it significantly influences management strategies. Accurate identification of BTB allows for conservative treatment options, such as the use of stents and ATT, which can effectively manage the condition without the need for more invasive surgical interventions[10]. In this case, despite multiple stent placements and ERCP procedures, the diagnosis remained unclear until CBD brushing during ERCP finally revealed M. tuberculosis. Biliary brushing during ERCP plays a pivotal role in diagnosing BTB. ERCP allows for the collection of biliary aspirates for AFB staining or culture, and FNA of adjacent lymph nodes if necessary. With the development of these strategies tissue diagnosis can be established based on suspicion of the disease, negating the need for a laparotomy in the diagnosis of hepatobiliary TB[5].
Routine liver function tests typically do not provide significant diagnostic insights for BTB, as they often remain within normal ranges[3]. However, imaging techniques can offer additional clues. Abdominal X-rays may reveal calcifications in about 50% of hepatic BTB cases, presenting as “chalky” and confluent calcifications along the course of the CBD[12]. Ultrasonography may show dilated intrahepatic ducts or CBD[3]. Pathological examination of bile duct specimens in BTB typically reveals multiple small cavities filled with greenish necrotic material, bile pigment, and caseation as in this patient whose pathological specimen showed multiple granulomas[9]. A valuable addition to the diagnostic toolkit in such cases is molecular diagnostics, such as polymerase chain reaction (PCR) for TB DNA. This technique can significantly improve the preoperative detection of M. tuberculosis, especially in smear-negative or paucibacillary cases, and may help avoid delays or unnecessary surgeries. Identifying TB DNA in blood and bile specimens through molecular assays before surgery can further aid in the preoperative diagnosis[10]. Preoperative diagnosis enables timely initiation of ATT and appropriate biliary decompression techniques, such as transhepatic biliary drainage or stenting, reducing the need for more invasive surgical procedures[10]. For patients with biliary stones, cholecystectomy combined with ATT is often required[13]. The standard treatment regimen for TB includes a combination of isoniazid, rifampin, ethambutol, and pyrazinamide, typically administered over a 6 to 9-month period.
Conclusion
Early and accurate diagnosis of BTB is vital for effective management and avoiding unnecessary surgical interventions. Considering TB in the differential diagnosis of biliary strictures, particularly in patients with a history of TB, is essential. Clinicians should maintain a high index of suspicion for BTB in cases of recurrent biliary obstruction with inconclusive biopsy or FNA results, especially when imaging mimics cholangiocarcinoma or sclerosing cholangitis.
Biliary brushing during ERCP should be prioritized as a critical diagnostic step in such cases, given its utility in detecting M. tuberculosis and avoiding diagnostic delays. Incorporating molecular assays such as PCR for TB DNA into diagnostic protocols may further improve early detection.
Future research should focus on the development of noninvasive diagnostic tools and biomarkers to identify BTB earlier in its course. Additionally, exploring the role of advanced imaging modalities—such as contrast-enhanced MRI or PET-CT—in differentiating TB-related strictures from malignant or autoimmune causes may provide diagnostic clarity and guide management.
Further research is needed to enhance our understanding of BTB and improve long-term management and prognosis[10].
Footnotes
Footnote Group
Contributor Information
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Ethical approval
Ethics approval was not required for this editorial.
Consent
Written informed consent was obtained from the patient for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
Source of funding
None to declare.
Conflicts of interest disclosure
The authors have no competing interests.
Guarantor
Muhammad Waqas Khan.
Provenance and peer review
Not commissioned, externally peer-reviewed.
Data availability statement
Not applicable.
References
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Associated Data
Data Availability Statement
Not applicable.