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  Table of Contents    
CASE REPORT  
Year : 2013  |  Volume : 56  |  Issue : 4  |  Page : 422-424
Epithelioid hemangioma of the testis


1 Department of Pathology, Shaoxing People's Hospital and Zhejiang University Shaoxing Hospital, Shanghai, China
2 Xin Hua Hospital affiliated to Shanghai JiaoTong University School of Medicine, Shanghai, China

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Date of Web Publication18-Jan-2014
 

   Abstract 

Testicular hemangioma is a very rare benign vascular neoplasm, there are only less than 30 cases of testicular hemangiomas reported in the literature. We report a case of epithelioid hemangioma of the testis in a 53-year-old man who presented with painless mass in the right testis. An orchiectomy was performed, The 1 cm tumor was composed of prominent proliferation of small, capillary-sized vessels lined by plump, epithelioid endothelial cells. Immunohistochemically, the epithelioid endothelial cells reacted with antibodies to factor VIII-related antigen, CD31, CD34 and Vimentin, but not CK, EMA, CEA, S-100 and CD68. Although it is a rare tumor, we should be aware of it and avoid confusion with other vascular neoplasm.

Keywords: Angiolymphoid hyperplasia with eosinophilia, epithelioid haemangioma, testis

How to cite this article:
Liu X, Wang R, Guan W, Wang L. Epithelioid hemangioma of the testis. Indian J Pathol Microbiol 2013;56:422-4

How to cite this URL:
Liu X, Wang R, Guan W, Wang L. Epithelioid hemangioma of the testis. Indian J Pathol Microbiol [serial online] 2013 [cited 2019 Dec 6];56:422-4. Available from: http://www.ijpmonline.org/text.asp?2013/56/4/422/125359



   Introduction Top


Epithelioid hemangioma, most frequently affected sites being the subcutis or dermis of the head and neck region, is a scarce type of vascular tumor of the testis. To review literature, only three cases related to epithelioid hemangioma of the testis were described as histiocytoid hemangioma or benign hemangioendothelioma, [1],[2],[3] which were proved to be hemangioendothelioma of the testis. Recently, three cases of epithelioid hemangioma of testis have been reported. [4] But it is unclear if this lesion has similar behavior and shares a common pathogenesis with superficial lesions. We describe a case of epithelioid hemangioma of the testis.


   Case Report Top


A 53-year-old man found a lump in his right scrotum himself 3 days earlier before hospitalization. Physical examination revealed a small walnut sized lump. Diagnosis was confirmed by ultrasonography of the scrotum, which also revealed a 0.9 cm diameter tumor of the right testis [Figure 1]. No other pathological condition was identified. The patient underwent orchiectomy. Examination of the specimen revealed a solid tumor of 1 cm in diameter with a dark red cut surface in the right testis. Histopathological examination of the excised specimen confirmed the diagnosis of epithelioid hemangioma. The patient is now in good general health after postoperative 6 months.
Figure 1: (a) Ultrasonography of the scrotum revealed a hypoechoic areas 0.9 cm in diameter lacking a clear boundary and capsule in the right testi s. (b) Color Doppler flow imaging indicated an blood fl ow in the tumor

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The tumor was composed of prominent proliferation of small, capillary-sized vessels lined by plump, epithelioid endothelial cells. The vessels typically have an immature appearance and they lack a well-defined lumen, but they are well formed with single cell layering of the endothelium. The endothelial cells have amphophilic or eosinophilic cytoplasm that is sometimes vacuolated, and they contain a single, relatively large, nucleus with an open chromatin pattern and a central nucleolus. An inflammatory milieu including a few eosinophils and lymphocytes is present in the case. The vascular origin of the tumor was demonstrated convincingly by immunohistochemical techniques with positive expressions of factor VIII-related antigen, cluster of differentiation 31 (CD31), cluster of differentiation 34 (CD34), and Vimentin, and negative expressions of cytokerain (CK), epithelial membrane antigen (EMA), prostate specific antigen (PSA), prostate-specific acid phosphatase (PSAP), carcinoembryonic antigen (CEA), S-100 protein(S-100) and cluster of differentiation 68 (CD68) for epithelioid endothelial cells [Figure 2].
Figure 2: Photomicrograph of epithelioid hemangioma of right testi s. (a) The tumor is located in right testis and characterized with diff use scatt er of small, capillary-sized vessels, HE ׃n40. (b) The vessels typically have an immature appearance and they lack a well-defi ned lumen, HE ׃n100.
(c) Plump and epithelioid endothelial cells have amphophilic or eosinophilic cytoplasm and a large nucleus with an open chromati n patt ern, HE ׃n 400. (d) The positi ve expression of CD31, IHC ׃n100. (e) The positi ve expression of factor VIII-related anti gen, IHC ׃n100. (f) The positi ve expression of CD34, IHC ׃n100


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   Discussion Top


The etiology of epithelioid hemangioma is still unknown. There remains considerable controversy as to whether epithelioid hemangioma is a reactive lesion or a true neoplasm. Epithelioid hemangioma is a relatively uncommon, benign vascular tumor that typically occurs in middle-aged women and the most frequently affected sites are the subcutis or dermis of the head and neck region. However, other rare sites including bone, lacrimal gland, inner canthus, and heart have been successively reported in the literature. [5],[6],[7],[8] The most frequent preoperative clinical impressions are an epidermal cyst or angioma. In the recent years, epithelioid hemangioma was suggested as an alternate term for "angiolymphoid hyperplasia with eosinophilia" to emphasize the characteristic epithelioid or histiocytoid morphology of the proliferating endothelial cells and to reflect the belief that this was a benign process.

The diagnosis was based on the typical histopathological features that show a benign vascular tumor with well formed but often immature vessels, the majority of which are lined by plump, epithelioid (histiocytoid) endothelial cells with amphophilic or eosinophilic cytoplasm, and a large nucleus with an open chromatin pattern and central nucleolus. It is common to encounter epithelioid endothelial cells within the lumen of the affected artery, either replacing part of the normal endothelial lining or "coating" fibrin fronds. Most cases have a prominent inflammatory component, but a notable eosinophilic reaction may not be essential to epithelioid hemangioma. [9] The epithelioid endothelial cells can be demonstrated convincingly by immunohistochemistry, which are immunoreactive for endothelial cell markers such as factor VIIIRAg, CD31 and CD34, and also positive to vimentin, which indicates a mesenchymal origination. The epithelioid tumor cells are negative to CK, PSA, PSAP, CEA, EMA, S-100 protein, and CD68, which is helpful for differential diagnosis.

The most important differential diagnosis of epthelioid hemangioma is Kimura's disease, which usually shows high eosinophils and IgE in peripheral blood. Histologycally, the Kimura's disease displayed hyperplasia of lymphoid tissue, with the formation of lymphoid follicles and active germinal centers, proliferation of post-capillary venules which were lined by plump endothelial cells. Another characteristic of the Kimura's disease is large number of eosinophils present around the vessels or adjacent to the follicles. In epithelioid hemangioma, however, lymphoid follicles were absent and eosinophil infiltration was not as prominent as in Kimura's disease. Once the vascular nature of epithelioid hemangioma of the testis is recognized, it is important to avoid confusion with epithelioid hemangioendothelioma, a borderline or low grade malignant vascular tumor with metastatic potential, which is also rare in the testis. [10],[11] The tumor is characterized by chains and cords of epithelioid endothelial cells distributed in a myxohyaline stroma. The cells have eosinophilic cytoplasm containing vacuoles that deform the cytoplasm (blister cells); some vacuoles contain fragmented erythrocytes. These features were lacking in our case of testicular epithelioid hemangioma.

Complete local excision and follow up are optimal management for epithelioid hemangioma. Local recurrence occurs in up to one third of patients but there are no reports of distant metastases. Our patient has been found with no recurrence or metastasis over 6 months of follow-up period.

 
   References Top

1.Mazzella FM, Sieber SC, Lopez V. Histiocytoid hemangioma of the testis: A case report. J Urol 1995;153:743-4.  Back to cited text no. 1
    
2.Banks ER, Mills SE. Histiocytoid (epithelioid) hemangioma of the testis. The so-called vascular variant of "adenomatoid tumor". Am J Surg Pathol 1990;14:584-9.  Back to cited text no. 2
    
3.Hargreaves HK, Scully RE, Richie JP. Benign hemangioendothelioma of the testis: Case report with electron microscopic documentation and review of the literature. Am J Clin Pathol 1982;77:637-42.  Back to cited text no. 3
    
4.Kryvenko ON, Epstein JI. Testicular hemangioma: A series of 8 cases. Am J Surg Pathol 2013;37:860-6.  Back to cited text no. 4
    
5.Madhuri V, Walter NM, Korula RJ, Nair S. Epithelioid haemangioma (benign haemangioendothelioma of bone: A case report. Indian J Cancer 1996;33:173-7.  Back to cited text no. 5
[PUBMED]    
6.Coombes AG, Manners RM, Ellison DW, Evans BT. Lacrimal gland epithelioid haemangioma. Br J Ophthalmol 1997;81:1020.  Back to cited text no. 6
    
7.Mariatos G, Gorgoulis VG, Laskaris G, Kittas C. Epithelioid haemangioma (angiolymphoid hyperplasia with eosinophilia) in the inner canthus. J Eur Acad Dermatol Venereol 2001;15:90-1.  Back to cited text no. 7
    
8.de Nictolis M, Brancorsini D, Goteri G, Prat J. Epithelioid haemangioma of the heart. Virchows Arch 1996;428:119-23.  Back to cited text no. 8
    
9.Kitamura H, Ito S, Kuwana N, Yutani C. Epithelioid hemangioma of the temporal artery clinically mimicking temporal arteritis. Pathol Int 1999;49:831-5.  Back to cited text no. 9
    
10.Enzinger FM, Weiss SW. Benign tumors and tumor-like lesions of blood vessels. In: Soft Tissue Tumors, 2 nd ed. St. Louis, MO: CV Mosby; 1988. p. 489-532.  Back to cited text no. 10
    
11.Tsolos C, Polychronidis A, Sivridis E, Kelidis G, Simopoulos C. Epithelioid hemangioendothelioma of the testis. J Urol 2001;166:1834.  Back to cited text no. 11
    

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Correspondence Address:
Lifeng Wang
Department of Pathology, Xin Hua Hospital affiliated to Shanghai JiaoTong University School of Medicine, Shanghai - 200 092
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.125359

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