Cavernous hemangioma of adult pancreas: A case report and literature review

Table 1 Previously reported pancreas hemangiomas in literature

No.	Year	Country	Author	Age (yr)/sex	Site/size (cm)	Preoperative diagnosis	Treatment	Follow up
1	1939	-	Ranstrom	61/F	Head/7	Found at autopsy	-
2	1960	France	Ringoir	-	-	-	Surgery
3	1961	France	Ringoir	71/F	Head/15	-	Retrocolic gastroenterostomy, vagotomy
4	1972	France	Colardyn	42/F	Body tail/-	-	Fat free diet, anticholinergic1
5	1985	France	Mangin	62/F	Head to tail/20	Multicystic heterogenous structure	Laparotomy with observation1
6	1991	Japan	Kobayashi	30/M	Head/20	Cavernous hemangioma suggested on US/MRI	Pancreatoduodenectomy
7	1991	Germany	Dageforde	79/F	Body tail/6		Observation1
8	2003	Taiwan	Chang	70/F	Body tail/4	Heterogenous hypervascular solid mass. Serous/cystic adenoma/adenocarcinoma?	Subtotal pancreatectomy	Healthy at 14 mo
9	2006	Austria	Plank	36/M	Head/3	Hypervascular mass. Nonfunctioning neuroendocrine tumor	Hemangioma suspected during laparotomy, confirmed by intraoperative US. Not resected1
10	2008	China	Xu	-	-	Three cases reported	-
11	2009	United States	Mundinger	45/F	Head/5.5	Hypodensity mass in CT. Benign (duplication cyst, paraganglioma, cystic GIST) cyst	Frozen section: benign cyst. PPPD performed
12	2010	-	Jarboui	60/F	-	-	-
13	2011	Israel	Weidenfeld	73/F	Head/5	Cyst with solid part	Whipple’s procedure
14	2011	Malaysia	Lee	49/F	Neck/5	Non-enhancing septated cyst. Mucious cyst with malignant features	Central partial pancreatectomy and gastrostomy	Symptom free at 6 mo
15	2012	Germany	Kersting	53/M	Head/8	Non-enhancing mass. Adenocarcinoma?	Extirpation of the tumor
16	2013	China	Zhi-hua	23/F	Head/5.4	Non enhancing multilocular cyst with fluid fluid levels	Subtotal pancreatectomy	Healthy, no recurrence at 1 yr
17	2013	United Kingdom	Malik	70/F	Head/8	Giant hemangioma suggested on CT	PPPD	Fine at 6 wk
18	2014	United Kingdom	Williamson	78/F	Head/4	Hemangioma per EUS	Observation1
19	2014	Japan	Naito	40/F	Body tail/10	Multilocular septated cystic mass	Pancreatectomy	No recurrence at 6 yr
20	2015	United States	Mondal	18/F	Head/6	Benign cyst	PPPD	Fine at 6 mo

¹Cases that were preoperatively suspected to be hemangioma and/conservatively managed. PPPD: Pylorus preserving pancreatoduodenectomy.

Table 2 Differential diagnoses of cystic and cystic-solid lesions of pancreas

Cystic lesions	Sex/age/size/site	Imaging, cytology features
Pseudocysts	Any	Unilocular
Serous cystic neoplasms	F/60s/large/body or tail	Serous and mucinous cystadenoma similar on CT
		Central fibrous scar with calcification
		Septation
		Multiple cysts lined by glycogen-rich epithelial cells that are positive for periodic acid Schiff and express cytokeratins
Mucinous cystic neoplasms	F/50-60s/> 10 cm/body or tail	Single multilocular cysts that do not communicate with the ductal system
		Smooth shape, even wall, with or without (fine) septa with potential small nodules
		Peripheral egg shell calcification
		Cyst lined by columnar mucin-producing epithelial cells set within an ovarian-like stroma
		Dysplasia and malignant potential
		Express keratin, carcinoembryonic antigen and CA19-9, while CK20 and CDX2 (markers of intestinal differentiation) are negative
IPMN	M = F/70-80s/head	Intraductal proliferation of mucinous cells usually showing papillary projections
		Dysplasia, invasion and malignant potential
		The epithelium of IPMNs expresses keratins, CEA and CA19-9, with variable expression of MUC
Solid pseudopapillary neoplasms	Young F/> 10 cm/tail, head	Solid or cystic-solid hypervascular tumors, solid part enhances
		Show pseudopapillae and pseudocysts
		Stain for vimentin and beta catenin with partial reactivity to keratin
		CD56 and synaptophysin stains may be positive, chromogranin negative
Cystic change of solid tumors	PDAC is rare < 50, M = F	Recognition of adjacent solid lesion (carcinoma) is the key to correct diagnosis of ductal adenocarcinoma
		PDAC are fibrotic, hypo-vascular, ill defined border, early infiltration in peri-pancreatic fat, invasion of vascular structures and ducts
		NET and Islet cell tumors are hyper-vascular and hyper-enhancing with positive staining for chromogranin and synaptophysin
Lymphoepithelial cysts	M/50-60s/ body or tail	Lined by squamous epithelium and surrounded by dense lymphoid tissue, possibly showing germinal centers
Vascular tumors (Lymphangiomas, hemangiomas, hemolymphangioma, hemangioendothelioma, angiosarcoma	F > M/large	Cystic-solid, encapsulated, multi-loculated, micro-cystic portions appear solid and enhancing
		Vascular markers CD31, CD34 and factor VIII are positive in endothelial cells
		Cytokeratins neg
		Lymphatic marker D2-40 neg in hemangioma, positive in lymphangioma
		Angiosarcomas, highly aggressive, a vascular channel lined by variably atypical endothelial cells
Metastatic from renal cell cancer		Hypervascular, invasive
Accessory splenic tissue	Tail	hypervascular
Others		Rare solid tumors of the pancreas also include: mature teratoma, hamartoma, sarcoidosis, yolk sac tumor, acinar cell carcinoma, lymphoplasmatic sclerosing pancreatitis, primary pancreatic lymphoma., duodenal tumor, duplication cysts, paraganglioma, cystic GIST, glomus tumor, etc.

IPMN: Intraductal papillary mucinous neoplasms; PDAC: Pancreatic ductal adenocarcinoma.