Atypical Biliary Adenofibroma of the Liver: Related Treatment and Performance

Abstract

Biliary adenofibroma is an extremely rare benign liver tumor, but it may be a precancerous lesion of cholangiocarcinoma. So far, only 29 cases have been reported in the literature. A 30-year-old woman was admitted to our department for upper abdomen mass. The computed tomography scan showed a huge cystic and partly substantial mass between the left lobe of the liver and the descending duodenum, which was considered to be an exophytic tumor derived from the left lobe of the liver. Laparoscopic liver segment IVb resection and cholecystectomy were performed. Microscopic examination showed that the tumor was composed of glandular cavities of varying sizes and fibrous interstitium. The glandular cavity was covered with cubic or columnar epithelium without atypia. Some of the mesenchymal cells are myofibroblast-like and spindle-shaped with red-stained cytoplasm. The mesenchymal cells in some areas proliferate densely with moderate atypia. It was considered to be an atypical biliary adenofibroma with focal necrosis and active cell proliferation which may have malignant transformation potential. There was no recurrence and metastasis at a 6-month follow-up. Biliary adenofibroma is a rare benign tumor derived from the bile duct, but it may progress to malignancy and develop distant metastasis. It is difficult to distinguish it from other liver tumors through imaging examination and the gold standard of diagnosis is histopathological examination. Close clinical follow-up is recommended.

Imaging Features of Biliary Adenofibroma With Malignant Transformation: A Case Report and Literature Review

“…Delayed enhancement of benign BAF may be related to a high content of fibrous stroma [1]. Among the cases that mentioned the DWI sequence, five showed obvious diffusion restriction, and the pathology suggested malignant transformation [6,11,13,16,17]. In summary, a lobulated and multiseptated cystic-solid mass located under the hepatic capsule, with the separation obviously enhanced, can help radiologists associate BAF.…”

“…More than half of the patients presented with epigastric pain, and 30% had no clinical symptoms. Three cases had an elevated CA19-9 concentration [3,5,6], and one case had an elevated CA12-5 concentration [7]. The other three cases had hepatitis B [8][9][10], and one case had hepatitis C [11].…”

“…Some lesions have obvious solid components, accompanied by enhanced arterial phase enhancement, of which nine cases described relatively low signal on the venous and delay phases. The pathological diagnosis showed malignant transformation in seven cases [3,6,10,11,13,16,17], which may be related to complex papillary, cribriformlike, back-to-back architecture, and less fibrous tissue. Delayed enhancement of benign BAF may be related to a high content of fibrous stroma [1].…”

“…We performed a detailed analysis of imaging information from previous cases (see Appendix) in which the median size of the lesion was 6.55 and ranged from 0.7 to 20 cm, which was mainly located under the capsule (50%) [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20]. Most of the lesions were multilobulated and multiseptated cystic.…”