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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 30  |  Issue : 3  |  Page : 321-323

Epithelioid hemangioma: A rare entity to report


Department of Oral Medicine and Radiology, VSPM Dental College and Research Centre, Nagpur, Maharashtra, India

Date of Submission05-Aug-2018
Date of Acceptance06-Aug-2018
Date of Web Publication18-Oct-2018

Correspondence Address:
Dr. Kunal Omprakash Sarate
Flat No. 403, Satyam Flora, Narendra Nagar, Nagpur, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_93_18

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   Abstract 


Epithelioid hemangioma (EH) or angiolymphoid hyperplasia is an uncommon benign vasculoproliferative neoplasm. We are reporting a case of EH involving the gingiva in a 12-year-old male patient who reported to the outpatient Department of Oral Medicine and Radiology with the chief complaint of gingival growth in the lower mandibular gingiva involving both buccal and lingual gingiva and facial asymmetry due to enlarged left submandibular lymph nodes. Histopathological features diagnosed the case definitive of EH.

Keywords: Epithelioid hemangioma, gingival growth, vasculoproliferative neoplasm


How to cite this article:
Motwani M, Karemore T, Sarate KO. Epithelioid hemangioma: A rare entity to report. J Indian Acad Oral Med Radiol 2018;30:321-3

How to cite this URL:
Motwani M, Karemore T, Sarate KO. Epithelioid hemangioma: A rare entity to report. J Indian Acad Oral Med Radiol [serial online] 2018 [cited 2019 Jan 23];30:321-3. Available from: http://www.jiaomr.in/text.asp?2018/30/3/321/243671




   Introduction Top


Epithelioid hemangioma (EH) is an uncommon benign vasculoproliferative neoplasm, whose etiology and pathogenesis are still uncertain. The term EH was coined by Enzinger and Weiss in 1983, but EH was first described in 1969 by Wells and Whimser as angiolymphoid hyperplasia with eosinophilia.[1],[2],[3]

Extraoral counterpart of this entity typically presents as a small angioma-like nodule, red to brown in color, which may be located intradermally or subcutaneously in young adults. It can mimic lymphoproliferative disorders, especially when the lesion arises in typical location, such as the extremities.[4] Oral presentation of EH is nonspecific and it may be difficult to clinically differentiate EH from angioma, pyogenic granuloma, Kaposi's sarcoma, salivary gland tumor, lymphoma, and squamous cell carcinoma.[1] Oral cases of EH were found to be common in males as compared with females, and the lips, tongue, buccal mucosa, and palate are the common sites involved.[5]

We are reporting a case of EH involving the gingiva in a 12-year-old male patient.


   Case Report Top


A 12-year-old male reported to the outpatient Department of Oral Medicine and Radiology with the chief complaint of gingival growth in the lower mandibular gingiva involving both buccal and lingual gingiva. Patient gave history of gradual increase in size and involvement with history of occasional gingival bleeding in the last 2 years. No history of weight loss or fever or other constitutional symptoms. Clinical examination showed granular growth involving marginal and attached gingiva, covering cervical third of all teeth in the region [Figure 1]. The growth was painless and not associated with bleeding on probing or any discharge. Lymph node examination revealed enlarged left submandibular node of size was 2 × 1.5 cm, oval shaped, firm, mobile, and nontender. In addition, two smaller submandibular lymph nodes, one on left side and one on right side, were also enlarged and nontender. Patients consent was obtained and radiological investigations such as anIntra oral periapical radiograph and Orthopantomograph were made, which revealed moderate alveolar bone resorption extending from 32 up to 37 region. On ultrasonography of the neck, except for enlarged lymph nodes, no other pathology was observed. Incisional biopsy was done, and at the same time, other investigations were carried out, such as Mantoux test, chest X–ray, and USG-guided FNAC for the lymph node to rule out any systemic cause. USG-guided FNAC for left submandibular enlarged lymph node suggested it as reactive lymphadenopathy, whereas other tests revealed no abnormality.
Figure 1: Extent of lesion from 32 up to the retromolar region involving both buccal and lingual gingiva

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Histopathologically, partly ulcerated stratified squamous epithelium showing focal acanthosis and papillomatosis was seen [Figure 2]. Subepithelial tissue showed lobules of small sized blood vessels lined by single layer of plump, epithelioid, endothelial cells with abundant eosinophilic cytoplasm, large nuclei, and central nucleoli [Figure 3]. The vessels were well canalized and red blood cells seen in the lumen. Few areas showed sheets of epithelioid epithelial cells. Intervening stroma shows mixed inflammatory infiltrate composed of mainly lymphocytes, few plasma cells, eosinophils, and occasional neutrophils, and areas of hemorrhage were also seen. Histopathologically features were suggestive of epithelioid hemangioma. Immunohistochemical evaluation with CD-34 marker heighted subepithelial nodular proliferation of numerous small blood vessels and features were consistent of capillary hemangioma [Figure 4], [Figure 5], [Figure 6]. The epithelioid hemangiomas are classified under capillary hemangiomas.[6]
Figure 2: IOPA showing moderate alveolar bone loss with 35, 36, and 37 regions

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Figure 3: OPG showing moderate alveolar bone loss extending from 32 up to 37 region

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Figure 4: Histopathological section showing partly ulcerated stratified squamous epithelium showing focal acanthosis and papillomatosis

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Figure 5: Histopathological section showing subepithelial tissue with blood vessels lined by epithelioid, endothelial cells with abundant eosinophilic cytoplasm

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Figure 6: Heighted subepithelial nodular proliferation of numerous small blood vessels with CD 34 Marker

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   Discussion Top


Routinely, hemangiomas of the oral soft tissues appear as painless, soft, smooth or lobulated, sessile, or pedunculated masses and may be seen in any size from a few millimeters to several centimeters.[7] EH may vary in clinical presentation depending on the site of involvement.

The exact etiology of EH is not known, but literature review suggests that EH can be a reactive condition caused by trauma, leading to arteriovenous malformation that may trigger cellular proliferation and growth secondary to damage or repair of an artery or vein.[5],[8] Present case does not report the relevance of trauma to the development of the lesion, but as suddenly developed lesion that progressed in size gradually. According to Henriques et al., literature has reported only 40 cases of EH involving the oral cavity. The mean age of the patients was 37.7 years, with a slight predilection for young adult men. Out of the 40 cases of EH reported, till date only one case was seen involving gingiva in a 30-year-old female.[1] Present case of 12-year-old male shows the extensive involvement of gingiva primarily. Lymph node enlargement was found to be reactive, which could be secondary to chronic nature of the lesion. This finding also favors the benign form of EH. Histopathology has been considered to be the gold standard for diagnosis of EH. Treatment of EH is often a challenge. Intralesional corticosteroids, cryotherapy, laser cauterization, and irradiation have been used but are not proven to be effective.[2],[9] Other therapeutic options available are intralesional interferon alpha-2a, indomethacin farnesil, pentoxifylline, and chemotherapeutic agents such as vinblastine, mepolizumab (anti-interleukine-5), and imiquimod.[9]

In one of the reported cases of EH involving lip, imiquimod 5%, receptor-7 agonist was given, which showed a successful resolution of the lesion without relapse in the follow-up period. Imiquimod enhances the innate and acquired immune response and also activates TH-1 cells, which provide a long-term memory of immune response to viral/tumor cells.[9] But, surgical excision with periodic follow-up has been accepted as recommended treatment till now. In the present case, patient was advised for surgical treatment. It is reported that patients with oral EH underwent for complete surgical resection without additional therapy, and recurrence was relatively rare in such cases.[7],[10],[11]


   Conclusion Top


This may be the second case of EH involving gingiva till now. EH can be presented in various forms like swelling or nodule, papule, salivary gland tumor, pyogenic granuloma, squamous cell carcinoma, and sometimes gingival enlargement; therefore, it is always recommended that it should be included in the differential diagnosis of similar soft tissue lesions.[12],[13],[14] Thorough clinical examination supplemented with histopathological analysis will help for definite diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Henriques ÁC, Vidal MT, Gurgel CA, Rocha SL, Júnior BC, de Azevedo RA, et al. Epithelioid hemangioma in the oral mucosa – A case report. Srp Arh Celok Lek 2016;144:535-40.  Back to cited text no. 1
    
2.
Kumari VR, Vallabhan CG, Geetha S, Nair MS, Jacob TV. Atypical presentation of capillary hemangioma in oral cavity – A case report. J Clin Diagn Res 2015;9:ZD26-8.  Back to cited text no. 2
    
3.
Nangia R, Puri A, Gupta R, Bansal S, Negi A, Chauhan I, et al. Epithelioid hemangioma of lingual alveolar mucosa: An immunohistochemical case report. Case Rep Med 2014;2014:436240.  Back to cited text no. 3
    
4.
Guo R, Gavino AC. Angiolymphoid hyperplasia with eosinophilia. Arch Pathol Lab Med 2015;139:683-6.  Back to cited text no. 4
    
5.
Aggarwal A, Keluskar V. Epithelioid hemangioma (angiolymphoid hyperplasia with eosinophilia) in the oral mucosa. Indian J Dent Res 2012;23:271-4.  Back to cited text no. 5
  [Full text]  
6.
Nayak VJ, Chatura RK, Morphological characterisation of capillary hemangioma pattern: An Indian study. Indian J Pathol Res Pract 2017;6:135-40.  Back to cited text no. 6
    
7.
Sun ZJ, Zhang L, Zhang WF, Alsharif MJ, Chen XM, Zhao YF, et al. Epithelioid hemangioma in the oral mucosa: A clinicopathological study of seven cases and review of the literature. Oral Oncol 2006;42:441-7.  Back to cited text no. 7
    
8.
Shimoyama T, Horie N, Ide F. Epithelioid hemangioma of the tongue mimicking a malignancy. J Oral Maxillofac Surg 2000;58:1317-9.  Back to cited text no. 8
    
9.
Venkatesan A, Singh R. Angiolymphoid hyperplasia with eosinophilia of oral mucosa in a child treated with imiquimod. Indian J Paediatr Dermatol 2016;17:196-8.  Back to cited text no. 9
  [Full text]  
10.
Miličć B, Velnar T, Pregelj R, Limbaeck-Stokin C. Multiple epithelioid hemangiomas with orbital involvement. Case Rep Pathol 2015;2015:629805.  Back to cited text no. 10
    
11.
Martín-Granizo R, Muñoz E, Naval L, Martín R, Goizueta C, Diaz FJ, et al. Epithelioid hemangiomas of the maxillofacial area. A report of three cases and a review of the literature. Int J Oral Maxillofac Surg 1997;26:212-4.  Back to cited text no. 11
    
12.
Danjanov I, Linder J. Soft Tissue Tumour, Anderson's Patholgy. 10th ed. St. Louis: Mosby; 1996. p. 2497.  Back to cited text no. 12
    
13.
Weiss SW, Goldblum JR. Benign tumours and tumour like lesions of blood vessels. Ezinger and Weiss's Soft Tissue Tumour. 4th ed. St. Louis: Mosby; 2001. p. 856-63.  Back to cited text no. 13
    
14.
Rosai J. Lymph nodes. Ackerman's Surgical Pathology. 7th ed. New Delhi: Jaypee; 1989. p. 856-63.  Back to cited text no. 14
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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