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

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Year : 2013  |  Volume : 17  |  Issue : 2  |  Page : 311-314

A rare case of angiolymphoid hyperplasia with eosinophilia in the submental region

1 Department of Oral Medicine and Radiology, Saraswati Dental College and Hospital, Juggour (via Chinhat), Lucknow, Uttar Pradesh, India
2 Department of Conservative and Endodontics, Saraswati Dental College and Hospital, Juggour (via Chinhat), Lucknow, Uttar Pradesh, India

Date of Web Publication11-Oct-2013

Correspondence Address:
Priya Singh
C-319, Nirala Nagar, Lucknow - 226020, Uttar Pradesh
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-029X.119757

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Angiolymphoid hyperplasia with eosinophilia is a rare reactive, angio-proliferative lesion which is usually found in the subcutaneous tissue of the head and neck. The lesion rarely arises from a native blood vessel. It is of unknown etiology but may be related to some benign, localized form of vasculitis. While preauricular location is the most frequent site of occurrence as reported in literature, a lesion in submental region seems to be a first reported site of occurrence. We report such an occurrence in an individual with history of trauma twice in his lifetime at the affected site. Spontaneous regression is common, but persistent or recurrent lesions may require treatment. Several treatments have been reported but surgery is considered to be the most efficient one.

Keywords: Angiolymphoid hyperplasia, eosinophilia, head and neck, lymphoid tissue

How to cite this article:
Singh P, Singh A. A rare case of angiolymphoid hyperplasia with eosinophilia in the submental region. J Oral Maxillofac Pathol 2013;17:311-4

How to cite this URL:
Singh P, Singh A. A rare case of angiolymphoid hyperplasia with eosinophilia in the submental region. J Oral Maxillofac Pathol [serial online] 2013 [cited 2023 Mar 28];17:311-4. Available from: https://www.jomfp.in/text.asp?2013/17/2/311/119757

   Introduction Top

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign but potentially disfiguring vascular lesion. It is usually characterized by dermal and subcutaneous nodules, primarily in the head and neck region and is especially common in preauricular region. Although rare, this entity should be considered in the differential diagnosis of hypervascular masses of the superficial head and neck region. We present a rare site of occurrence of angiolymphoid hyperplasia with eosinophilia in the submental region. [1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13]

   Case Report Top

A 22-year-old man reported with a complaint of a hard disfiguring swelling in the chin region. The swelling had gradually increased in size to attain the present size and had hardened in consistency to attain the present consistency. Patient had first noticed the swelling 1΍ years back and complained of mild associated pruritis. The patient gave a history of trauma to the same region twice, the first about 10 years ago, which was associated with significant bleeding and had required suturing and the second about 2 years ago. His medical history was non-contributory. He had no tissue abuse or tobacco abuse habit.

On extra-oral examination, a single, well-defined, non-tender nodular swelling measuring about 2.5 cm Χ 1.5 cm in size with firm to hard consistency was present in the submental region [Figure 1]. The nodule was movable in nature. The temperature over the nodule was normal. Intra-orally there was no abnormality detected apart from moderate gingival recession with lower left central incisor.
Figure 1: Clinical picture showing extraoral nodular swelling at the submental region

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Periapical, occlusal and panoramic radiographs were obtained and were non-contributory. The laboratory investigations that were advised for the patient included complete blood count, bleeding time, clotting time, random blood sugar, hepatitis B surface antigen test and human immunodeficiency virus test. All the reports were within the normal limits except for differential leukocyte count, which showed marked increase in eosinophils to 8% (normal 1-6%). The lesion was surgically excised and sent for histopathological examination [Figure 2].
Figure 2: The excised specimen in toto obtained from the submental region

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   Histopathology Top

The tissue section showed:
  • Vascular proliferation [Figure 3]
  • Lymphoid proliferation [Figure 4]
  • Vascular proliferation with thickened blood vessel walls showing large plump endothelial cells having oval nuclei with vacuolated cytoplasm [Figure 5]
  • Dense stroma with inflammatory cells viz. abundant eosinophils with central necrosed areas and lymphocytes [Figure 6].
Figure 3: Vascular proliferation (H&E stain, ×100)

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Figure 4: Photomicrograph showing lymphoid proliferation (H&E stain, ×100)

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Figure 5: Vascular proliferation with thickened blood vessel walls showing large plump endothelial cells having oval nuclei with vacuolated cytoplasm (H&E stain, ×400)

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Figure 6: Dense stroma having abundant eosinophils (H&E stain, ×400)

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   Differential Diagnoses Top

  • Angiolymphoid hyperplasia with eosinophilia
  • Kimura's disease
  • Cutaneous lymphoma
  • Cavernous hemangioma
  • Pyogenic granuloma
  • Kaposi's sarcoma
  • Bacillary angiomatosis. [13]

Cutaneous lymphoma

  • Epidermotropism
  • The intraepidermal cluster of lymphocytes (Pautrier's abscess)
  • Angiodestruction and tissue infarction
  • Infiltration into the subcutaneous fat (panniculitis-like cutaneous lymphoma) or blood vessels (intravascular lymphoma). [11]

Kimura's disease

  • Produces large subcutaneous nodules in the head and neck region with normal overlying skin
  • Often involves regional lymph nodes
  • Is a chronic inflammatory condition
  • Lacks "epithelioid" blood vessels
  • Elevated serum immunoglobulin E. [11],[14],[15],[16],[17],[18],[19],[20]

Angiolymphoid hyperplasia with eosinophilia

  • Proliferation of small blood vessels many of which are lined by enlarged endothelial cells with uniform ovoid nuclei and intra-cytoplasmic vacuoles (cobblestone appearance)
  • Perivascular and interstitial infiltrate composed primarily of lymphocytes and eosinophils (5-15% of infiltrate)
  • Lymphoid aggregates with or without follicle formation. [1],[4],[5],[6],[7],[8],[9],[10],[12],[16],[17],[18],[21],[22],[23]

Cavernous hemangioma

  • Large dilated blood sinuses with thin walls, showing endothelial lining
  • Sinusoidal spaces usually are filled with blood
  • Lymphatic vessels occur in some instances. [2],[11]

Pyogenic granuloma

  • Lobular pattern of vascular proliferation with inflammation and edema similar to granulation tissue
  • Thin and atrophic epidermis at the top with variable ulcerations
  • Vast numbers of endothelium-lined vascular spaces
  • Acanthosis and hyperkeratosis at sides
  • Extreme proliferation of fibroblasts and budding endothelial cells
  • Variable mitotic activity. [11]

Kaposi's sarcoma

  • Proliferation of small veins and capillaries around one or more preexisting dilated vessels
  • Slit-like vascular channels without a visible endothelial lining, typically interspersed with the spindle cells
  • Vessels are lined by plump, mildly atypical endothelial cells
  • Pronounced mononuclear inflammatory cells infiltrate, including mast cells, scattered erythrocytes and hemosiderin deposits may be present
  • Variable mitotic activity. [2],[3],[11]

Bacillary angiomatosis

  • The architecture resembles that of lobular capillary hemangioma, but the endothelial cells are often larger, polygonal and sometimes markedly atypical
  • There is presence of neutrophils, leukocytoclastic debris and granular material (bacteria). [11]

   Final Diagnosis Top

Angiolymphoid hyperplasia with eosinophilia.

   References Top

1.Nawaf AM, Manchanda Y. Angiolymphoid hyperplasia with eosinophilia associated with hepatitis C antibodies. Indian J Dermatol Venereol Leprol 2007;73:367.  Back to cited text no. 1
2.Fernandes BF, Al-Mujaini A, Petrogiannis-Haliotis T, Al-Kandari A, Arthurs B, Burnier MN Jr. Epithelioid hemangioma (angiolymphoid hyperplasia with eosinophilia) of the orbit: A case report. J Med Case Rep 2007;1:30.  Back to cited text no. 2
3.Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 1969;81:1-14.  Back to cited text no. 3
4.Karabudak O, Taskapan O, Bozdogan O, Dogan B. Angiolymphoid hyperplasia with eosinophilia: Atypical appearance in an older patient. Indian J Dermatol 2008;53:144-5.  Back to cited text no. 4
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5.Banerjee S. Angiolymphoid hyperplasia with eosinophilia. Indian J Dermatol 2005;50:238-9.  Back to cited text no. 5
6.Zaraa I, Mlika M, Chouk S, Chelly I, Mokni M, Zitouna M, et al. Angiolymphoid hyperplasia with eosinophilia: A study of 7 cases. Dermatol Online J 2011;17:1.  Back to cited text no. 6
7.Cornelius RS, Biddinger PW, Gluckman JL. Angiolymphoid hyperplasia with eosinophilia of the head and neck. AJNR Am J Neuroradiol 1995;16:916-8.  Back to cited text no. 7
8.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. 8
9.Moran CA, Suster S. Angiolymphoid hyperplasia with eosinophilia (epithelioid hemangioma) of the lung: A clinicopathologic and immunohistochemical study of two cases. Am J Clin Pathol 2005;123:762-5.  Back to cited text no. 9
10.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. 10
11.Pai K, Gupta A. Intravascular epithelioid haemangioma of temporal artery: A diagnostic difficulty. J Clin Diagn Res 2007;1:177-80.  Back to cited text no. 11
12.Olsen TG, Helwig EB. Angiolymphoid hyperplasia with eosinophilia. A clinicopathologic study of 116 patients. J Am Acad Dermatol 1985;12:781-96.  Back to cited text no. 12
13.Peters E, Altini M, Kola AH. Oral angiolymphoid hyperplasia with eosinophilia. Oral Surg Oral Med Oral Pathol 1986;61:73-9.  Back to cited text no. 13
14.Ahn HJ, Lee KG. A clinicopathological study of Kimura's disease and epithelioid hemangioma. Yonsei Med J 1990;31:205-11.  Back to cited text no. 14
15.Urabe A, Tsuneyoshi M, Enjoji M. Epithelioid hemangioma versus Kimura's disease. A comparative clinicopathologic study. Am J Surg Pathol 1987;11:758-66.  Back to cited text no. 15
16.Ko KF. Kimura's disease of the orbit. J Hong Kong Med Assoc 1990;42:227-8.  Back to cited text no. 16
17.Shetty AK, Beaty MW, McGuirt WF Jr, Woods CR, Givner LB. Kimura's disease: A diagnostic challenge. Pediatrics 2002;110:e39.  Back to cited text no. 17
18.Chong WS, Thomas A, Goh CL. Kimura's disease and angiolymphoid hyperplasia with eosinophilia: Two disease entities in the same patient: Case report and review of the literature. Int J Dermatol 2006;45:139-45.  Back to cited text no. 18
19.Ceçen E, Kaçar-Döger F, Etensel B. An extremely rare cause of generalized lymphadenopathy in children: Kimura's disease. Turk J Pediatr 2010;52:534-7.  Back to cited text no. 19
20.Calli C, Oncel S, Pinar E, Bener S, Calli A. Kimura's disease: Two case reports with a review of the literature. Kulak Burun Bogaz Ihtis Derg 2004;12:139-43.  Back to cited text no. 20
21.Shenefelt PD, Rinker M, Caradonna S. A case of angiolymphoid hyperplasia with eosinophilia treated with intralesional interferon alfa-2a. Arch Dermatol 2000;136:837-9.  Back to cited text no. 21
22.Andreae J, Galle C, Magdorf K, Staab D, Meyer L, Goldman M, et al. Severe atherosclerosis of the aorta and development of peripheral T-cell lymphoma in an adolescent with angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 2005;152:1033-8.  Back to cited text no. 22
23.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. 23


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

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