Abstract
We herein report the clinicopathologic features of a rare case of biliary adenofibroma (BAF) of the liver in a 79-year-old man. Grossly, tumour presented as a well-circumscribed, 5.5-cm mass with a solid and microcystic appearance. Histological examination was typical of biliary adenofibroma, showing a proliferation of variable-sized tubulocystic structures embedded in a moderately cellular fibrous stroma. Immunohistochemistry, revealing immunoreactivity of the epithelial component to cytokeratins 7 and 19, was consistent with a bile duct origin. Notably, the stromal cells had a myofibroblastic profile, showing a diffuse and strong expression of vimentin and α-smooth muscle actin. Differential diagnosis with Von Meyenburg complex, biliary adenoma, biliary cistadenoma, congenital biliary cystsy, and hepatic benign cystic mesothelioma is provided. The occasionally reported expression of p53 in biliary adenofibroma has suggested that this tumour could represent a premalignant lesion. The absence of both cytological atypia and p53 immunoreactivity in our case confirms that BAF is a benign tumour with an indolent clinical behaviour. However, a careful histological examination of BAF is mandatory because malignant transformation of the epithelial component has been documented in two cases.
Highlights
Benign biliary tumours are uncommon, including bile duct adenoma, biliary hamartoma, biliary cystadenoma, and the solitary bile cysts
biliary adenofibroma (BAF) is a benign tumour with a clinical indolent behavior, malignant transformation of the epithelial component [2, 3] with associated distant metastases [3] has been documented
BAF is a rare tumour of bile duct origin with only six cases reported in the literature to date [1,2,3,4,5,6] (Table 1)
Summary
Benign biliary tumours are uncommon, including bile duct adenoma ( known as peribiliary gland hamartoma), biliary hamartoma (von Meyenburg complex), biliary cystadenoma, and the solitary bile cysts. Tsui et al in 1993 described a new liver tumour entity called “biliary adenofibroma” (BAF) [1]. To the best of our knowledge, only six cases of BAF of the liver have been reported in the literature to date (Table 1) [1,2,3,4,5,6]. Monosomy 22, a cytogenetic alteration found in some benign mesenchymal neoplasms, has been documented in one case of BAF [5]. BAF is a benign tumour with a clinical indolent behavior, malignant transformation of the epithelial component [2, 3] with associated distant metastases [3] has been documented
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