Intrahepatic biliary cystadenoma: A case report

Table 1 Literature review for the clinical features of biliary cystadenoma

Ref.	Case number	Mean age (yr)	Sex	Clinical manifestation	CA199 (iu/mL)	Treatment
Lewis et al[9], 1988	15	41	F (13), M (2)	Abdominal pain (12), sepsis/cholangitis (4), lower-extremity edema (2), abdominal mass (2), no symptom (2)	NA	Left lateral segmentectomy (2), right trisegmentectomy (1), liver transplant (1), wedge resection of cyst (2), central hepatic resection (1), left trisegmentectomy (2), right hepatic lobectomy (2), marsupialization of cyst (1), hepatic cystectomy (3)
Stoupis et al[3], 1994	1	37	F	Jaundice, pruritus	NA	Mass resection
Hara et al[11], 2001	1	48	F	No symptom	Normal	Left hepatectomy
Kazama et al[6], 2005	1	68	M	Abdominal fullness, nausea, vomiting	NA	Right hepatic lobectomy
Lewin et al[15], 2006	6	55	F (6)	Abdominal pain (3), jaundice (2), no apparent symptom (1)	Normal (6)	Mass resection (6)
Yu et al[8], 2008	2	63.5	M (2)	Abdominal pain (2)	107 (1), normal (1)	Left lobectomy plus cholecystectomy (1), resection was not performed due to the metastatic disease
Delis et al[5], 2008	4	51	F (4)	Abdominal pain, occasionally vomiting	Increased (4)	Left hepatectomy (3), right hepatectomy and left hepaticojejunostomy (1)
Romagnoli et al[13], 2011	1	53	F	Obstructive jaundice	Normal	Liver transplant
Gonzalez et al[1], 2009	1	32	F	Abdominal pain, jaundice, diarrhoea	43	Left hepatectomy
Sang et al[10], 2011	19	44.2	F (17), M (2)	Abdominal pain (9), vomiting (1), palpable abdominal mass (1), jaundice (1), no symptom (10)	Increased (9)	Cyst enucleation (3), laparoscopic fenestration (1), right hemihepatectomy (1), mentectomy (2), left lateral sectionectomy (2), left medial sectionectomy (1), right segmentectomy (1), right anterior sectionectomy (1), right posterior sectionectomy (1)
Williamson et al[7], 2013	3	45.3	F (3)	No symptom (1), abdominal pain (1), epigastric mass (1), hepatic cyst infection (1)	NA (1), increased (2)	Wedge resection (1), surgery (1), left hemihepatectomy (1)
Yang et al[4], 2013	1	57	F	Intermittent abdominal pain	Normal	Left hepatic lobectomy
Liu et al[2], 2018	1	59	F	Jaundice, palpable abdominal mass	Increased	Left hemihepatectomy
This case	1	58	M	Abdominal fullness	Normal	Left hepatic lobectomy, cholecystectomy, liver cyst fenestration

Normal < 35 iu/mL. F: Female; M: Male; NA: Not available.

Table 2 Literature review for imaging features of biliary cystadenoma

Ref.	Location	Imaging findings (US, CT, MRI, MRCP)
Lewis et al[9], 1988	Left lobe (5), right lobe (8), porta hepatis (1), central (2)	US demonstrated fluid-filled lesions with internal septation and nodularity consistent with the papillary infoldings. CT revealed a smooth, thick walled cyst with thin internal septations
Stoupis et al[3], 1994	Right lobe	CT showed a large septate, hypoattenuating mass with enhancement of the septations. MRI showed multilocular lesion with different signal intensities in the locules
Hara et al[11], 2001	Left lobe	CT demonstrated a multiple low density mass. MRI showed multilocular cysts, with homogeneous signals on T1WI and T2WI
Kazama et al[6], 2005	Right lobe	CT demonstrated a giant, well-circumscribed, low-density mass with internal septa
Lewin et al[15], 2006	Left lobe	MRI revealed a homogeneous cystic lesion in the six BCAs. Three BCAs showed a hypointensity on T1WI and an isointensity in the other three. A fluid/fluid level consistent with internal hemorrhage was found in one BCA. MRCP showed ductal dilatation upstream to cystic lesion in two BCAs
Yu et al[8], 2008	Left lobe	MRI showed a cystic mass in the left hepatic lobe. MRCP showed diffuse dilatation of the biliary tree with different signals in the bile ducts
	Left lobe	CT revealed a large cystic mass with septations and multiple papillary projections
Delis et al[5], 2008	Left lobe (3), right lobe (1)	US revealed various sizes of cysts with septations and infolding projections originating from the cystic wall. CT revealed cysts with internal septations and mural nodules with contrast enhancement
Romagnoli et al[13], 2011	Segments 4 and 8	CT and MRI revealed a cystic mass with septations in its caudal portion, lying on the Glissonian bifurcation, causing dislocation of the portahepatis and compression of the main right and left hepatic ducts with peripheral bile duct dilatation
Gonzalez et al[1], 2009	Left lobe	CT showed a large water density mass with internal septa and without calcifications
Sang et al[10], 2011		CT and MRI revealed fluid-containing, well-defined, lobulated, multilocular, thick walled cystic masses with enhanced internal septation and convex papillae
Williamson et al[7], 2013	Left lobe (2), central lobe (1)	CT confirmed a large hepatic cyst with calcification. MRI showed a cystic lesion containing proteinaceous fluid or blood, which had a solid component with papillary projections
Yang et al[4], 2013	Left lobe	CT revealed a hypoattenuating shadow with internal septae and enhanced after intravenous administration of contrast medium
Li et al[2], 2018	Left lobe	CT revealed a hypoattenuating shadow with slight enhanced internal septations. Fluid on MRI revealed a low signal intensity on T1WI and high signal intensity on T2WI without enhancement
This case		CT revealed multiple sizes of hypoattenuating shadows with nodular calcification and internal septations and slight enhancement. MRI showed that internal septation separated the fluid-filled spaces and was enhanced

US: Ultrasound; CT: Computed tomography; MRI: Magnetic resonance imaging; MRCP: Magnetic resonance cholangiopancreatography.