Intrahepatic multicystic biliary hamartoma: A case report

Table 1 Summary of patients with multicystic biliary hamartoma in the world literature

Case No.	Ref.	Patients ’age (years)/Sex	Location	Size (cm)	Imaging or histological features	Treatment
1	Kobayashi et al[4], 2005	30/M	Seg VI	3.6	Embedded in a fibrous stroma and lined by low columnar or cuboidal epithelium	Partial resection
2	Zen et al[5], 2006	59/M	Seg IV	4.2	A relatively well-circumscribed nodule was enhanced on CT by contrast medium and sustained until the delayed phase	Left hepatectomy
3		70/F	Seg III	1.8	A relatively well-circumscribed nodule	Segmentectomy
4		69/F	Seg III	2.8	Showed a multilocular cystic lesion containing many small cystic spaces	Segmentectomy
5	Kai et al[6], 2008	55/M	Seg VI	5.0	Abdominal magnetic resonance imaging revealed as a low density area on T1-weighted images and a multiple bulboid high intensity area on T2-weighted images, respectively	Partial resection
6	Ryu et al[7], 2010	45/M	Seg VII	2.0-3.5 (case nos. 6-8)	CT during arterial portography (CTAP) also shows that normal liver parenchyma can be found around the cystic lesions	Partial resection
7		58/M	Seg III		CT (precontrast) shows multiple cysts and a small calcification	Partial resection
8		55/F	Seg VI,VII			Partial resection
9	Song et al[2], 2013	52/M	Seg III	2.7	On T2-weighted MRI, conglomerated multiple cystic nodules with a high signal intensity were seen. Thin septae and the wall of the lesion were enhanced on contrast-enhanced, T1-weighted MRI	Partial resection
10	Beard et al[8], 2014	48/F	Seg VII	4.7	Microscopic exam demonstrated thick, dense fibrous tissue containing cytologically bland, large caliber bile ducts with intermingled benign hepatocytes	Extended right hepatectomy
11	Yoh et al[9], 2014	69/M	Seg III	3.0	The peripheral site of this lesion is slightly enhanced on the arterial phase. On the portal phase, the ring-enhancement of the lesion is clearer and shows honeycomb-like dilated bile duct	Left hepatectomy
12	Fernández-Carrión et al[10], 2014	60/F	Seg VI	5.0		Partial resection
13	Tominaga et al[11], 2015	26/M	Seg V,VI	10.0	Histologically, these cystic lesions were composed of variably and irregularly dilated duct structures lined by columnar epithelium resembling bile duct lining. There were no atypical cells and no papillary growth of the epithelial cells	Right hepatectomy
14	Morinaga et al[12], 2017	53/M	left lobe of the liver	12.0	Enhanced abdominal computed tomography and magnetic resonance imaging (MRI) revealed a multicystic tumor with a calcified wall in the left lobe of the liver	Left hepatectomy
15	Ogura et al[13], 2018	77/F	Seg III	12.0	CT demonstrates a huge, low-density lesion with strong enhancement at the periphery	Partial resection
16	Mu et al[14], 2021	37/M	Seg VI	8.0	Calcification was observed in dilated ducts; The enhancement of the septa and peripheral wall within the cystic lesion (arterial phase); The lesion showed a low-density honeycomb-like appearance (venous phase)	Laparoscopic partial resection
Presentcase		14/M	Seg III	17.0	A large, cystic, space-occupying lesion, with blurred outline with respect to adjacent liver structures	Laparoscopic partial resection

M: Male; F: Female; CT: Computed tomography; MRI: Magnetic resonance imaging.