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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
CASE REPORT Table of Contents   
Year : 2005  |  Volume : 9  |  Issue : 1  |  Page : 21-23
 

Epithelioid hemangioma of the alveolar mucosa


1 Department of Oral and Maxillo Facial Pathology, Ragas Dental College and Hospital, Chennai, India
2 Department of Conservative Dentistry, Ragas Dental College and Hospital, Chennai, India

Correspondence Address:
T R Saraswathi
Department of Oral and Maxillo Facial Pathology, Ragas Dental College and Hospital, 2/102, East coast road, Uthandi, Chennai
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.39055

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   Abstract 

Epithelioid hemangioma (EH) is a benign vascular lesion composed primarily of epithelioid endothelial cells accompanied by a chronic inflammatory infiltrate. This lesion is also known as Histiocytoid hemangioma or Angiolymphoid hyperplasia with eosinophilia (ALHE). This rare tumour arising from vascular structures has uncertain etiology. It often occurs in the skin and subcutaneous fat of the head and neck region, but may also arise in the deep tissues such as muscle, bone and lymph node. The intraoral lesions of EH are uncommon and local recurrence has been observed only in a few cases. We report a case of epithelioid hemangioma involving the alveolar mucosa with local recurrence.


Keywords: Epithelioid hemangioma, vascular, histiocytoid, recurrence


How to cite this article:
Saraswathi T R, Nalin Kumar S, Jayanthi P, Umadevi M, Ranganathan K, Srinivasan M R. Epithelioid hemangioma of the alveolar mucosa. J Oral Maxillofac Pathol 2005;9:21-3

How to cite this URL:
Saraswathi T R, Nalin Kumar S, Jayanthi P, Umadevi M, Ranganathan K, Srinivasan M R. Epithelioid hemangioma of the alveolar mucosa. J Oral Maxillofac Pathol [serial online] 2005 [cited 2019 Sep 19];9:21-3. Available from: http://www.jomfp.in/text.asp?2005/9/1/21/39055



   Introduction Top


Epithelioid hemangioma (EH) is a rare lesion, which often occurs in the skin and subcutaneous tissues of the head and neck region [1] . It was first reported as Angiolymphoid hyperplasia with eosinophilia (ALHE) by Wells & Whimster in 1969 [2] and Histiocytoid hemangioma by Rosai et al in 1979 [3] . The term Epithelioid hemangioma was coined by Enzinger and Weiss in 1983 [4] to describe this benign vascular lesion [2] . Infra oral lesions of EH are uncommon and only 16 cases have been reported in the English literature. We report a case of epithelioid hemangioma involving the alveolar mucosa in a 68-year-old female patient with local recurrence.


   Case Report Top


A 68-year-old female reported to the out patient department of Ragas Dental College & Hospital for the complaint of a painful swelling in the upper left posterior region of one month duration. History revealed that, since the onset of the swelling, there was no alteration in size. The medical and dental history of the patient was non-contributory. The patient did not give any history of trauma to the involved area.

Extra oral examination of the head and neck did not show any abnormalities. On intraoral examination, a tender, soft, friable swelling measuring 1 x 0.5 sq. cm was present in the alveolar mucosa of 26, 27 region. The lesion was erythematous and the tooth in relation to the mass revealed no pathology. The intraoral periapical radiographs and OPG revealed no abnormalities. The patient was not diabetic, not hypertensive and the routine hematological findings were within normal limit. A provisional clinical diagnosis of reactivc lesion was made.

Excisional biopsy of the lesion was done under local anesthesia. Microscopic examination of the biopsy specimen revealed that the lesion was composed of vascular and inflammatory cell components. The vascular component consisted of marked proliferation of endothelial­ lined blood vessels [Figure - 1]. The proliferating endothelial cells had a predominantly epithelioid appearance [Figure - 2]. In many foci, the endothelial cells were plump and projected into the lumen giving the "tomb stone" appearance [Figure - 3],[Figure - 4]. Intracytoplasmic vacuoles were seen in some foci [Figure - 5]. A dense mixed inflammatory cell infiltrate comprising predominantly of eosinophils and lymphocytes was also seen [Figure - 6]. A diagnosis of epithelioid hemangioma was made based on the histopathologic features.

The patient was advised to come fix a regular follow up. Two months after the excisional biopsy, the patient reported back with a similar lesion in the same site. Surgical excision of the recurrent mass was done and the tissue was subjected to histopathological examination. The recurrent lesion also revealed features of epithelioid hemangioma.


   Discussion Top


EH is all uncommon lesion seen more often in Asians and Caucasians [6] . It is present most commonly in patients aged 20-50 years, with a mean onset of 30-33 years. This condition is rare in elderly patients and in non-Asian paedriatic population [6] . EH is somewhat store common in females. The case reported here was found in 65-year-old patient, which is uncommon. The extra oral sites of EH are periauricular region [1] . forehead, and scalp. Pruritis is the most common presenting complaint [6] . Hew cases with multiple lesions have been reported [7] . In 20% of the cases, the lesion is associated with lymphadenopathy and blood eosinophilia [7] .

Although EH has been considered to be a benign tumor in literature: numerous factors suggest that it may occur as a result of an unusual reactive process [6],[7] . Fetsh and Weiss reviewed 96 cases of EH. out of which 87% showed vascular damage in the histopathological features Thcy considered that EH is not neoplastic, but suggested an arteriovenous malformation resulting from trauma as the precursor to EH and it arises in a reactive basis secondary to damage and repair of an artery or vein (2). But, in our ease the patient could not recall any previous injury to the involved area. EH was suggested to be a hypersensitivity response by some authors based on the eosinophilia seen in some patients [6] . In our patient, the absolute eosinophil count was within normal limits ruling out this possibility. The other suggested causes of EH are low-grade infection and hormonal imbalance [7]

Histologically, EH exhibits prominent proliferation of vessels lined by plump "epithelioid" endothelial cells, with abundant eosinophilic cytoplasm and central oval nucleus [1],[5] . Intracytoplasmic vacuoles can he identified in sonic cells [3] . The stroma of the tumour is funned by fibromyxoid tissue with inflammatory cells that surround the vessels [1] . The inflammatory cell infiltrate is composed of eosinophils, plasma cells, lymphocytes, macrophages, and mast cells [3] .

lmmunohistochermical analysis reveal a positive reaction of EH for Vimentin and factor VIII related antigen. EH exhibits a negative reaction for other markers such as cytokeratin, EM A, S-100, desmin and smooth muscle actin [5] .

Although there has been a controversy concerning the relation between FH and Kimura's disease, recent studies indicate that the two diseases are different entities [3] . In Kimura's disease, (issue blood eosinophilia is commonly seen and the absence of epithelioid endothelial cells is another demarcating feature [1],[2] .

The differential diagnosis of EH also includes other epithelioid vascular tumours and non-vascular soft tissue tumours showing epithelioid characteristics. Among the family of epithelioid vascular tumours, epithelioid angiosarcoma is the differential diagnosis in the malignant end with epithelioid hemangioendotheloima occupying the borderline malignant position [8] . Angiosarcoma of the oral mucosa is rare and is an infiltrative destructive tumor composed of markedly pleomorphic cells and numerous abnormal mitoses. Oral epithelioid hemangioendothelioma is composed of mildly pleomorphie epithelioid endothelial cells, but intravascular papillary growth and myxohyaline stroma are the diagnostic clues [5] . The other distinguishing histopathologic features of hemangioendothelioma arc cellular atypia, mitosis and presence of necrosis [7] .

Other microscopically similar soft tissue tumours such as epithelioid neurofibroma, epithelioid schwannoma, epithelioid fibrosarcorna may be considered, but their histological appearance is sufficiently distinctive to allow the differentiation [5] . The other differential diagnoses of EH are pyogenic granuloma, eosinophilie granuloma and pseudolymphoma [6] .

Local recurrence has been observed in about 20% of the reported cases with oral lesion [5] . Our patient had presented with a recurrent lesion 2 months after the initial excision.

Once the diagnosis is confirmed, surgical resection [1],[3] with conservative margins is the treatment although superficial irradiation, cryotherapye and intralesional steroid therapy [3] have been used in few patients [6] . The purpose of this article is that EH should he in the differential diagnosis of a peripheral intraoral lesion in elderly patients.

 
   References Top

1.Kurihara K, Saiki T, Takeda K, Kohayashi J. Epithelioid Hemangioma of the Maxillary Sinus: A Case Report. J Oral Maxillofac Surg 1995: 53: 1221-1223.  Back to cited text no. 1    
2.Granizo RM, Munoz E, Naval L., Martin R. Goizueta C, Diaz FJ. Epithelioid Hemangioma of the Maxillofacial area: A Report of three cases and a review of the Literature. Int J Oral Maxillofac Surg 1997; 26: 212-214.  Back to cited text no. 2    
3.Toeg A, Kermish M, Grishkan A, Temkin D. Histiocytoid hemangioma of the oral cavity: A Report of two cases, J Oral Maxillofac Surg 1993;51:812-814.  Back to cited text no. 3    
4.Enzinger FM, Weiss SW. Soft tissue tumours. 3 rd edition: Mosby St. Louis: 593 -600  Back to cited text no. 4    
5.Shimoyama T, Horie N, Ide F. Epithelioid Hemangioma of the Tongue Mimicking a Malignancy. J Oral Maxillofac Surg 2000; 58: 1317-1319.  Back to cited text no. 5  [PUBMED]  [FULLTEXT]
6.Rajendran R, Padmakumar SK, Balaraman Nair M. Angiolymphoid Hyperplasia with Eosinophilia: A Case Report. JIDA 2003; 74: 605-608.  Back to cited text no. 6    
7.Marrogi AJ, Boyd D, EL-Mofty SK, Waldron C. Epithelioid Hemangioendothelioma of the oral cavity: Report of two cases and Review of Literature, J Oral Maxillofac Surg 1991; 49: 633­-638.  Back to cited text no. 7    
8.Orsini G, Fioroni M, Rubini C. Piattelli A. Epithelioid Hemangioendothelioma of the oral cavity: Report of case. J Oral Maxillofac Surg 2001;59: 334-337.  Back to cited text no. 8    


    Figures

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



 

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