Double Trouble: A Rare Case of Concurrent Biliary Adenofibroma and Hepatobiliary Mucinous Cystic Neoplasm

Abstract

Biliary adenofibroma and hepatobiliary mucinous cystic neoplasm are exceedingly rare tumors. To our knowledge, no case of co-existent biliary adenofibroma and mucinous cystic neoplasm has been previously reported. We present a patient who was diagnosed with both tumors concurrently. Both can present with abdominal pain, although some are diagnosed incidentally in asymptomatic patients. Imaging and tumor markers can be suggestive, but histologic examination is needed for definitive diagnosis. Both have a propensity for malignant transformation, thus complete surgical resection is the treatment of choice. Although rare, awareness of these tumors leads to earlier diagnosis and treatment.

Imaging presentation of biliary adenofibroma: A case report

“…Currently, although BFs are classified as benign bile duct tumors and precursors by the 2019 World Health Organization tumor classification system[ 5 ], some cases of BF are characterized by malignant transformation, invasion, and even distant metastasis (Tsui et al [ 1 ] first proposed in 1993, postoperative pathology showed malignant transformation in 3 cases[ 6 - 8 ], 3 cases of biliary adenofibroma with invasive carcinoma[ 9 - 11 ], 3 cases were complicated with liver tissue invasion[ 12 - 14 ], 2 cases were complicated with lymph node metastasis[ 15 ], local recurrence occurred in 2 cases[ 16 ]). The clinical manifestations and imaging characteristics of BF are nonspecific.…”

“…Including the present case, 25 cases of BF have been reported to date (Tsui et al [ 1 ] in 1993, Parada et al [ 17 ] in 1997, Haberal et al [ 12 ] in 2001, Akin et al [ 18 ] and Garduño-López et al [ 19 ] in 2002, Gurrera et al [ 20 ] in 2010, Kai et al [ 6 ] and Nguyen et al [ 7 ] in 2012, Jacobs et al [ 13 ] in 2015, Godambe et al [ 9 ]; Thai et al [ 10 ] and Thompson et al [ 15 ] in 2016, Kaminsky et al [ 8 ] and Arnason et al [ 16 ] in 2017, Esteban et al [ 11 ] in 2018, Sturm et al [ 14 ] in 2019), and no differences in sex or age at onset are evident. The mean age at onset is 57.2 ± 17.79 years.…”

Magnetic Resonance Imaging Findings of Biliary Adenofibroma

“…thus far in the medical literature. 1,[3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19] Previously reported cases focused on the clinical and pathological features of biliary adenofibroma, while the imaging findings were recorded only sporadically. Currently, MRI is the imaging modality of choice for evaluating a range of intrahepatic tumors.…”