Solitary hepatic lymphangioma mimicking liver malignancy: A case report and literature review

Table 1 Basic characteristics and manifestations of reviewed solitary hepatic lymphangioma

No.	Ref.	Year	Age	Sex	Manifestation
1	Kohashi et al[32]	2017	50 yr	F	Epigastric discomfort
2	Lee et al[14]	2016	42 yr	F	Right upper abdominal pain
3	Zouari et al[21]	2015	8 mo	M	Abdominal distention
4	Nakano et al[19]	2015	18 yr	F	Epigastric pain
5	Liu et al[22]	2014	41 yr	M	Right upper abdominal pain
6	Zhang et al[4]	2013	42 yr	F	None
7	Nazarewski et al[10]	2013	41 yr	F	None
8	Peña Ros et al[17]	2013	20 yr	F	Abdominal pain
9	Ak et al[18]	2012	58 yr	F	Weight loss
10	Huang et al[23]	2010	35 yr	M	Right upper abdominal pain
11	Matsumoto et al[1]	2010	52 yr	F	Upper abdominal pain
12	Shahi et al[24]	2009	22 d	M	Abdominal distension
13	Zeidan et al[25]	2008	23 yr	M	Abdominal pain or vomiting
14	Stunell et al[36]	2009	30 yr	F	Right upper quadrant pain
15	Nzegwu et al[26]	2007	4 mo	M	Abdominal distension
16	Chan et al[28]	2005	15 yr	F	Upper abdominal pain
17	Koh et al[29]	2000	6 mo	M	Abdominal girth
18	Stavropoulos et al[20]	1994	64 yr	M	Epigastric pain
19	Clerici et al[27]	1989	ND	F	ND

ND: Not described.

Table 2 Imaging of reviewed solitary hepatic lymphangioma

No.	Ultrasound	CT	MRI
1	Low-echoic, unilocular cystic mass	Low density, non-enhancement	ND
2	Mixed-echoic, mass with cystic and solid components	Low density, non-enhancement	ND
3	Unilocular cystic mass	Low density, non-enhancement	ND
4	Mixed-echoic, mass with cystic and solid components	Low density, non-enhancement	Mixed signal
5	Mixed-echoic, mass with cystic and solid components	Low density, heterogeneous enhancement	ND
6	Mixed-echoic, mass with cystic and solid components	Low density, non-enhancement for cystic region, enhancement for septa	ND
7	Non-echoic, multilocular cystic mass	Low density, non-enhancement for cystic region, enhancement for septa	Enhancement for septa on T1
8	ND	Low density, non-enhancement	ND
9	ND	Low density, non-enhancement for cystic region, enhancement for septa	Low signal on T1, high signal on T2
10	Non-echoic, multilocular cystic mass	Heterogeneous density	Mixed signal on T2
11	Mixed-echoic, mass with cystic and solid components	Low density, non-enhancement for cystic region, enhancement for septa	Low signal on T1, high signal on T2
12	Unilocular cystic mass	Low density	ND
13	Multilocular cystic mass	ND	Low signal, enhancement for septa on T1
14	Non-echoic, multilocular cystic mass	Low density, non-enhancement for cystic region, enhancement for septa	ND
15	Multilocular cystic mass	ND	ND
16	ND	Low density, non-enhancement for cystic region, enhancement for septa	ND
17	Multilocular cystic mass	Low density	ND
18	Mixed-echoic, mass with cystic and solid components	Low density	ND
19	Unilocular cystic mass	Low density	ND

CT: Computed tomography; MRI: Magnetic resonance imaging; ND: Not described.

Table 3 Initial diagnosis and surgical findings of reviewed cases

No.	Initial diagnosis	Location	Number	Size in cm	Texture	Type of surgery
1	Hepatic cyst	Hepatoduodenal ligament	1	13 × 10	Unilocular cyst with serous fluid	LPR/CR
2	Hepatic cystadenoma/cystadenocarcinoma	Segments 7 and 8	1	23 × 30	Multilocular cyst with bloody fluid	CR
3	ND	Right lobe	1	14.9 × 10.7 × 9.7	Unilocular cyst	CR
4	Rupture of a hepatic tumor	Left lobe	1	12.0 × 9.5	Multilocular cyst with blood clot	CR
5	Bile duct cyst/biliary cystadenoma/cystadenocarcinoma/hydatid cyst	Segment 2	1	16.2 × 13.0 × 4.3	Multilocular cyst with serous fluid	CR
6	Hepatic hemangioma/cystadenocarcinoma	Right lobe	1	23 × 15 × 7	Multilocular cyst with brownish fluid	CR
7	Hepatic cyst	Gallbladder and hepatoduodenal ligament	1	13.0 × 5.5 × 6.5	Multilocular cyst with serous fluid	CR
8	ND	Segment 5	1	15 × 9	Unilocular cyst	CR
9	Liver malignancy	Segment 5	1	3.0 × 2.2 × 1.5	Multilocular cyst with mucoid fluid	CR
10	Cystadenoma/cystadenocarcinoma/hepatic cyst with bleeding	Segments 4, 5, 6, 7, 8	1	25 × 23	Multilocular cyst with blood clot	CR
11	Sclerosing hemangioma/biliary cystadenoma/cystadenocarcinoma	Segment 4	1	3	Multilocular cyst with mucoid fluid and solid content	CR
12	Uncertainty	Right lobe	1	9.6 × 8.0 × 13.0	Unilocular cyst with yellowish fluid	CR
13	ND	Right lobe	1	12.6 × 9.4 × 11.5	Multilocular cyst with serous fluid	CR
14	Hepatic cyst	Gallbladder	1	15.2 × 9.4	Multilocular cyst with serous fluid	CR
15	Hepatic cyst	Right lobe	1	ND	Multilocular cyst with serous fluid	CR
16	Hepatic lymphangioma	Segments 4 5, 6	1	21 × 20 × 6	Multilocular cyst with brownish fluid	CR
17	Lymphangioma/mesenteric cyst	Segment 4	1	20 × 14	Multilocular cyst	CR
18	Uncertainty	Segment 3	1	4	Sponge-like cyst with chylous fluid and blood	CR
19	Hydatid cyst	ND	1	ND	Cystic	Resection

CR: Complete resection; LPR: Laparoscopic partial resection; ND: Not described.

Table 4 Histology and follow-up of reviewed solitary hepatic lymphangioma

No.	Histology	VWF	CD-31	CD-34	D2-40	LYVE-1	Prox-1	Discharged in d	Follow-up in mo	Prognosis
1	Cystic lymphangioma	ND	ND	-	+	ND	ND	ND	24	Alive/disease free
2	Cystic lymphangioma	ND	+	ND	ND	ND	ND	10	18	Alive/disease free
3	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	ND	ND
4	Cystic lymphangioma	ND	+	ND	+	ND	ND	8	48	Alive/disease free
5	Cystic lymphangioma	ND	ND	-	ND	ND	ND	ND	30	Alive/disease free
6	Cystic lymphangioma	ND	ND	+	-	ND	ND	ND	24	Alive/disease free
7	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	ND	Alive/disease free
8	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	ND	ND
9	Cavernous lymphangioma	ND	ND	ND	ND	ND	ND	ND	ND	ND
10	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	17	Alive/disease free
11	Cystic lymphangioma	-	+	+	+	+	+	ND	6	Alive/disease free
12	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	5	1	Alive/disease free
13	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	24	Alive/disease free
14	Cystic lymphangioma	ND	+	ND	ND	ND	ND	4	ND	ND
15	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	4	Alive/disease free
16	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	18	Alive/disease free
17	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	3	Alive/disease free
18	Cavernous lymphangioma	ND	ND	ND	ND	ND	ND	7	24	Alive/disease free
19	Cystic lymphangioma	ND	ND	ND	ND	ND	ND	ND	ND	ND

-: Negative; +: Positive. ND: Not described; VWF: Von Willebrand factor.