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


 
  Table of Contents    
CASE REPORT  
Year : 2015  |  Volume : 19  |  Issue : 1  |  Page : 108
 

Angiolymphoid hyperplasia with eosinophilia in the angle region of the mandible


1 Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, YüzüncüYil University, Van, Turkey
2 Department of Pathology, Gulhane Military Medical Academy, Ankara, Turkey

Date of Submission11-Jun-2014
Date of Acceptance20-Mar-2015
Date of Web Publication19-May-2015

Correspondence Address:
Dr. Cennet Neslihan Eroglu
Yuzuncu Yil Universitesi, Dis Hekimligi Fakultesi, Kampus, Van - 65080
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.157216

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   Abstract 

Angiolymphoid hyperplasia with eosinophilia (ALHE), also called epithelioid hemangioma, is a rare benign vascular lesion usually affecting the muscular arteries of the head and neck in female patients. Here, we report a 30-year-old male patient who presented with painless swelling in the angle region of the mandible. The diagnosis of the specimen, which was surgically removed under local anesthesia, was made as ALHE. The patient has remained uneventful for 3 years.


Keywords: Angiolymphoid hyperplasia, angle of mandible, eosinophilia, epithelioid hemangioma


How to cite this article:
Ozkan BT, Eroglu CN, Cigerim L, Gunhan O. Angiolymphoid hyperplasia with eosinophilia in the angle region of the mandible. J Oral Maxillofac Pathol 2015;19:108

How to cite this URL:
Ozkan BT, Eroglu CN, Cigerim L, Gunhan O. Angiolymphoid hyperplasia with eosinophilia in the angle region of the mandible. J Oral Maxillofac Pathol [serial online] 2015 [cited 2021 Mar 1];19:108. Available from: https://www.jomfp.in/text.asp?2015/19/1/108/157216



   Introduction Top


Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign vascular lesion. [1] The most frequently involved regions are the head and neck, hands, penis and arms. [2],[3],[4]

ALHE typically manifests with single or multiple, red/brown dome-shaped papule or subcutaneous nodules. In some cases, nodules extend into the dermis or muscular layer. Peripheral eosinophilia and lymphadenopathy are observed in 20% of patients. [5],[6],[7] The etiology of ALHE remains indefinite, but it may be vascular. [6],[8]

ALHE is more prevalent in Caucasian women aged 30-40 years. [9] Histologically, it appears as reactive proliferation of a small blood vessel that surrounds a muscle artery together with inflammatory infiltrates. [6]


   Case report Top


A 30-year-old male patient presented to the Department of Maxillofacial Surgery with swelling in the right facial region. His anamnesis revealed that the patient had his right first molar tooth extracted owing to infection a year ago and that painless swelling in that region had been present ever since. Clinical examination of the swelling demonstrated a painless, hard, mobile, localized elastic lesion on palpation in the subcutaneous tissues extending in line with mandibular border. The lesion was located approximately between the angle of the mandible and the anterior aspect of the sternocleidomastoid muscle (2-3 cm below the inferior border of the mandible) [Figure 1]a].
Figure 1: (a) Preoperative patient's extraoral view. (b) Intraoperative view of the lesion. (c) Resected surgical specimen. (d) 12th month Postoperative extraoral view

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There was no lymphadenopathy in the region. No specific finding was detected on radiological examination. The lesion was explored at the level of the subplatismal plane via extraoral approach under local anesthesia through an incision made parallel to the mandibular border. The lesion, which appeared of vascular origin because of red-brown color, was capsulated and associated with a small artery on both the anterior and posterior borders [Figure 1]b].

The borders of the lesion were ligated with polyglycolic acid suture to control bleeding . After total enucleation, subcutaneous tissues were sutured with 3/0 polyglycolic acid and the skin was sutured with 4/0 polyprolene. The excised specimen measuring 1.5 cm was sent to the laboratory for histopathological examination [Figure 1]c].

Histopathological examination revealed a restricted tumoral lesion consisting of vascular configurations of various diameters. Vascular configurations were rich with capillary vessels arranged in a lobular pattern. There was a large blood vessel with patent lumen and thick wall in the middle of the lesion [Figure 2]a].
Figure 2: (a) Histopathological image of the large blood vessel (H&E stain, x40). (b) Photomicrograph showing lymphocyte and eosinophilic leukocyte infiltration along with blood capillaries (H&E stain, x200)

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Vascular configurations were lined by epithelioid endothelial cells. Endothelial cells showed no atypia. Edematous connective tissue with lymphocyte and eosinophilic leukocyte infiltration were detected among vascular configurations [Figure 2]b].

Under the light of these findings, the patient was diagnosed with epithelioid hemangioma. No esthetic problem or complaint or recurrence was encountered over the course of the 3-year follow-up period [Figure 1]d].


   Discussion Top


Here, we report a case of ALHE in the angle region of the mandible in a 30-year-old male patient. The etiology of ALHE remains unknown, because it is not clear if it is primarily a vascular neoplasm, a lymphoproliferative process or a heterogeneous group of entities. Trauma, infections and renin or hyperestrogenic conditions (pregnancy or oral contraceptive agents) are considered to be the likely causes. [10],[11] Infection was considered as the etiological factor in the present case report, because it was learned from his anamnesis that swelling caused by an infected first molar tooth a year ago induced the pathology in that region.

ALHE usually appears in head and neck region, frequently in the auricular area and usually measures about 2-3 cm in size. However, other authors have reported that lips, oral mucosa and tongue may also be involved. [3],[12] In the present case, epithelioid hemangioma involved the angle region of the mandible. In this respect, the present case report might contribute to the literature of other cases of epithelioid hemangioma that might be presented in the future.

In the literature, it is usually mentioned that ALHE is an asymptomatic lesion, more prevalent among females and that lymphadenopathy is present in about 5-20% of patients. [12] Asymptomatic disease course may sometimes become symptomatic because of occasional recurrence of inflammation. We believe that lymphadenopathy might be detected in that period. Particularly, considering that ALHE is a reactive lesion, frequent palpation of the lesion by the patients may be triggering the inflammatory process.

ALHE must be histologically and clinically differentiated from Kimura disease, which is a chronic inflammatory condition characterized by large subcutaneous nodules in the head and neck region. It should be kept in mind that Kimura disease is more prevalent among young males; it is associated with increased immunoglobulin E levels and skeletal involvement may be encountered. Peripheral eosinophilia and lymphadenopathy may be encountered in both diseases. The most significant distinctive criterion is the fact that whilst ALHE is pathology of vascular origin, Kimura disease is a chronic inflammatory process. [10],[12]

In addition to Kimura disease, differential diagnosis of ALHE includes cutaneous lymphoma, cavernous hemangioma, pyogenic granuloma, Kaposi sarcoma and bacillary angiomatosis. [13]

Vascular proliferation in subcutaneous tissue, the presence of cobblestone-like endothelial cells lining the vessels, lymphocyte infiltration together with eosinophilia are important histopathological criteria for diagnosis. [14] These features were key in the diagnosis of the present case. Eosinophilic chemotactic factor released from mast cells is suggested as an extra agent contributing to eosinophilia, which adds to the inflammatory characteristics of such cases. Eosinophil counts typically return to normal after complete remission of the lesion. [15] Surgical excision is the preferred method for the treatment of ALHE. Alternative therapies include electrodessication, curettage, radiotherapy, cryotherapy, chemotherapy, corticosteroids, laser surgery and various agents, for example, interferon alpha 2b. [7] Spontaneous remission in such cases is possible within months, even years, but recurrences are frequent. Treatment is necessary in symptomatic cases and in situations that alter the patient's appearance. [10] In the present case, we preferred surgical excision for esthetic concern. We believe that complete surgical resection is a successful therapeutic option for such cases, because of its feasibility and practicality.



 
   References Top

1.
Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 1969;81:1-14.  Back to cited text no. 1
    
2.
Abrahamson TG, Davis DA. Angiolymphoid hyperplasia with eosinophilia responsive to pulsed dye laser. J Am AcadDermatol 2003;49:S195-6.  Back to cited text no. 2
    
3.
Park Y, Chung J, Cho CG. Angiolymphoid hyperplasia with eosinophilia of the tongue: Report of a case and review of the literature. Oral Oncol 2002;38:103-6.  Back to cited text no. 3
    
4.
Ozcanli H, OzenciAM, Ozcanli C, Ibis S, Gurer IE. Angiolymphoid hyperplasia: A case of a rare arterial involvement and successful recurrence treatment with laser therapy. J Eur Acad Dermatol Venereol 2007;21:1106-7.  Back to cited text no. 4
    
5.
Kempf W, Haeffner AC, Zepter K, Sander CA, Flaig MJ, Mueller B, et al. Angiolymphoid hyperplasia with eosinophilia: Evidence for a T-cell lymphoproliferative origin. Hum Pathol 2002;33:1023-9.  Back to cited text no. 5
    
6.
Gonzalez-Cuyar LF, Tavora F, Zhao XF, Wang G, Auerbach A, Aguilera N, et al. Angiolymphoid hyperplasia with eosinophilia developing in a patient with history of peripheral T-cell lymphoma: Evidence for multicentric T-cell lymphoproliferative process. Diagn Pathol 2008;3:22.  Back to cited text no. 6
    
7.
Alcántara González J, Boixeda P, TruchueloDíez MT, Pérez Garcia B, JaénOlasolo P. Angiolymphoid hyperplasia with eosinophilia treated with vascular laser. Lasers Med Sci 2011;26:285-90.  Back to cited text no. 7
    
8.
Vadlamudi G, Schinella R. Traumatic pseudoaneurysm: A possible early lesion in the spectrum of epithelioid hemangioma/angiolymphoidhypreplasia with eosinophilia. Am J Dermatopathol 1998;20:113-7.  Back to cited text no. 8
    
9.
Jeon EK, Cho AY, Kim MY, Lee Y, Seo YJ, Park JK, et al. Angiolymphoid hyperplasia with eosinophilia that was possibly induced by vaccination in a child. Ann Dermatol 2009;21:71-4.  Back to cited text no. 9
    
10.
Kaur T, Sandhu K, Gupta S, Kanwar AJ, Kumar B. Treatment of angiolymphoid hyperplasia with eosinophilia with the carbon dioxide laser. J Dermatolog Treat 2004;15:328-30.  Back to cited text no. 10
    
11.
Miller CJ, Ioffreda MD, Ammirati CT. Mohs micrographic surgery for angiolymphoid hyperplasia with eosinophilia. Dermatol Surg 2004;30:1169-73.  Back to cited text no. 11
    
12.
El Sayed F, Dhaybi R, Ammoury A, Chababi M. Angiolymphoid hyperplasia with eosinophilia: Efficacy of isotretinoin? Head Face Med 2006;2:32.  Back to cited text no. 12
    
13.
Singh P, Singh A. A rare case of angiolymphoid hyperplasia with eosinophilia in the submental region. J Oral Maxillofac Pathol 2013;17:311-4.  Back to cited text no. 13
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14.
Jang KA, Lee JY, Kim CH, Choi JH, Sung KJ, Moon KC, et al. Angiolymphoid hyperplasia with eosinophilia and Kimura's disease: Aclinico-pathologic study in Korea. Korean J Dermatol 2001;39:309-17.  Back to cited text no. 14
    
15.
Rosai J. Angiolymphoid hyperplasia with eosinophilia of the skin. Its nosological position in the spectrum of histiocytoid hemangioma. Am J Dermatopathol 1982;4:175-84.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2]



 

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