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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

Priya Singh¹, Abhinav Singh^2
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

Correspondence Address:
Priya Singh
C-319, Nirala Nagar, Lucknow - 226020, Uttar Pradesh
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/0973-029X.119757

Abstract

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

Introduction

Case Report

Figure 1: Clinical picture showing extraoral nodular swelling at the submental region Click here to view

Figure 2: The excised specimen in toto obtained from the submental region Click here to view

Histopathology

• 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) Click here to view

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

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

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

Differential Diagnoses

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

• 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]

• 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]}

• 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]}

• 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]

• 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]

• 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]

• 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

References

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