Case of a Rare Inflammatory Hepatic Hilar Mass Mimicking CholangiocarcinomaStephanie Egge MSIII, Debdeep Banerjee MSIII, Subhasis Misra M.D., H. Nail Aydin M.D.

Department of Surgery

Texas Tech University Health Sciences Center, Amarillo

References:1.Vasiliadis, K. et al. (2014). Mid common bile duct inflammatory pseudotumor mimicking cholangiocarcinoma. A case report and literature review. International Journal of Surgery Case Reports, 5(1), 12–15. 2.Worley, P. et al. (2001). Benign inflammatory pseudotumour of the biliary tract masquerading as a Klatskin tumour. HPB : The Official Journal of the International Hepato Pancreato Biliary Association  , 3(2), 179–181. 3.Dumitrascu, T. et al. (2009). Klatskin-mimicking lesions--a case series and literature review. Hepato-gastroenterology, 57(101), 961-967.4.Juntermanns, B et al. (2011). Klatskin-Mimicking Lesions: Still a Diagnostical and Therapeutical Dilemma?.5.Knoefel, W. T. et al. (2003). Klatskin tumors and Klatskin mimicking lesions of the biliary tree. European Journal of Surgical Oncology (EJSO), 29(8), 658-661.6.Kapoor, S., & Nundy, S. (2012). Bile duct leaks from the intrahepatic biliary tree: a review of its etiology, incidence, and management. HPB Surgery, 2012.

Discussion

•66-y/o healthy Hispanic male with 3-week •pruritic jaundice•nausea/vomiting, steatorrhea•18-lb weight loss over the prior 1 month•α-fetoprotein tumor marker (AFP) of 2.1 mM (normal: 0.5-2.5)

•Past Medical History: (1) Alcohol use disorder with 2 year sobriety (2) Cholecystectomy 2 years prior•Family History: Non-contributory•Social History: 30 year history of 12-18 12 oz. beers. Denies tobacco or illicit drug use or history.

Table 1. Liver Function Tests on Admission

Imaging Studies•CT abdomen: Heterogeneous hilar mass with intrahepatic biliary dilatation (Figure 1).•MRCP confirmed dilatation of intrahepatic ducts involving the confluence (see Figure 2).•ERCP with brush cytology with 10-Fr stent placement at the common hepatic duct bifurcation.

•Filling studies suggested defect at common hepatic duct, extending to proper hepatic branches

PathologyBrush histopathology: benign epithelium with evidence of chronic inflammationCT & US-biopsy: findings consistent with chronic inflammationDue to minimal extend of biopsies and the clinical picture (i.e. presence of a mass in the hepatic hilum), surgical exploration was pursued.

Surgical exploration: Diagnostic laparoscopy showed no evidence of peritoneal carcinomatosis. Open exploration demonstrated multiple lymphadenopathies of the hepatoduodenal ligament. Frozen sections showed findings consistent with chronic inflammation and histiocytosis.

Portal dissection revealed diffuse fibrotic and desmoplastic reactions, involving extrahepatic biliary ductal system and porta hepatis. Fibrosis and chronic inflammation persisted within the intrahepatic parenchyma, without local mass. Intraoperative-US and full-thickness biopsy were performed at the area of concern, revealing findings of chronic inflammation.

Based on histopathological evaluation and in the absence of malignancy., the case was terminated. Patient recovered unremarkably and has improved ever since with outpatient follow-up with gastroenterology and surgery.

Case Report

Hepatic hilar masses can be of several different etiologies, most commonly malignant. We present a rare inflammatory hepatic hilar lesion that mimics cholangiocarcinoma. Preoperative distinctions between benign, inflammatory hyperplasias and cholangiocarcinoma pose a challenge. Benign lesions mimicking cholangiocarcinoma most commonly auto-immunogenic or infectious etiologies, such as primary sclerosing cholangitis and recurrent pyogenic cholangitis. Benign and malignant hilar masses usually present with nonspecific symptoms of painless jaundice, weight loss, fatigue, and malaise. Radiologic techniques are often inadequate to detect major differences in morphology, and image-guided biopsy lacks adequate sensitivity to rule-out cholangiocarcinoma in the setting of benign disease. Biopsy-negative lesions are often presumed as cholangiocarcinoma and treated with extensive resections, and definitive exclusion of cholangiocarcinoma usually results from post-resection histopathological analysis. Here, we report a case of a rare inflammatory hepatic hilar mass, mimicking cholangiocarcinoma.

Introduction

Benign inflammatory lesions represent a rare etiology of obstructive hilar masses; however, diagnostic exclusion requires adequate preoperative and intraoperative work-up. Appropriate histopathological analysis includes frozen sectioning of hepatic lesion and exploration and biopsy of portal lymph nodes. If malignancy is evident, resection is indicated. However, in cases of benign histopathology, other potential etiologies such as infections (particularly viral), autoimmune, and allergic reactions should be explored. In such cases, non-operative management and further work up may be more appropriate, thereby avoiding potential morbidity and complications associated with hepatobiliary resection.

Conclusion

Value a

Alkaline phosphatase (ALP) [U/L] 856 ⇑ 44-147

Aspartate aminotransferase (AST) [U/L] 104 ⇑ 10-40

Alanine transaminase (ALT) [U/L] 66 ⇑ 7-56

Total bilirubin [mg/dL] 5.2/ ⇑ 0.3-1.9

Albumin [g/dL] 2.3 ⇓ 3.5-5.5

Value Normal Range

Alkaline phosphatase (ALP) [U/L] 302 ⇑ 44-147

Aspartate aminotransferase (AST) [U/L] 33.0 10-40

Alanine transaminase (ALT) [U/L] 32.0 7-56

Total bilirubin [mg/dL] 0.8 0.3-1.9

Albumin [g/dL] 3.4 3.5-5.5

Figure 1. CT-abdomen revealed a hilar liver mass and intrahepatic biliary dilation, suggestive of Klatskin tumor. Arrows denoting dilation. Arrow

head denoting heterogeneous enhancement.

Figure 2. MRCP showed intrahepatic ductal dilatation as far as the confluence of the intrahepatic ducts. MRI weighting is T2 in depicted images.

Recent patient data is provided in Table 2, supporting physiologic improvement. Recent EGD also revealed no new lymphadenopathies, inflammation, or mass that would be suspected with cancer.

A

B