Angiolymphoid hyperplasia with eosinophilia in the angle region of the mandible

Angiolymphoid Hyperplasia with eosinophilia (ALHE) is considered a rare, benign vascular lesion that mainly affects the skin and subcutaneous tissues of the head and neck, but is uncommon in the oral cavity. Its etiology remains unclear and it has been described as a reactive vascular proliferation, vascular malformation or neoplasm. Kimura' s disease is the primary entity to consider in differential diagnosis. Here we report on a rare case of ALHE involving the upper lip of a 50-year-old male patient that had a nodular swelling with approximately 3 cm, 7 years after initial onset. An excisional biopsy was performed and histopathologic examination revealed a well-encapsulated, multi-lobed lesion with proliferation of blood capillaries, displaying endothelial cells of epithelioid appearance, diffuse inflammatory infiltrate with lymphocytes, plasma cells, numerous eosinophils, and presence of lymphoid follicles. Immunohistochemical tests were positive for the markers CD34 and Ki-67 that, in combination with the results of morphological examination, were suggestive of a diagnosis of ALHE.Keywords: vascular system lesions; hemangioma; differential diagnosis. ResumoA hiperplasia angiolinfoide com eosinofilia (HALE) é considerada uma lesão vascular benigna rara que acomete, principalmente, o tecido cutâneo e subcutâneo da região de cabeça e pescoço, mas incomum na cavidade oral. Sua etiopatogenia permanece indefinida, sendo descrita como proliferação vascular reacional, malformação vascular ou neoplasia. Tem como principal diagnóstico diferencial a doença de Kimura. Este trabalho relata um caso de um paciente do sexo masculino, de 50 anos, que exibia aumento de volume nodular na mucosa do lábio superior, com 3 cm de dimensão e 7 anos de evolução. Após a biópsia excisional, o exame histopatológico mostrou lesão bem encapsulada multilobulada com proliferação de capilares sanguíneos com células endoteliais de aspecto epitelioide, infiltrado inflamatório difuso com linfócitos, plasmócitos, inúmeros eosinófilos e presença de folículos linfoides. A análise imuno-histoquímica revelou positividade para CD34 e Ki-67, o que, juntamente com o exame morfológico, direcionou o diagnóstico para HALE.Palavras-chave: lesões do sistema vascular; hemangioma; diagnóstico diferencial.

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“…11 In the present case, there was immunopositivity for CD-34, which agrees with findings in the literature.…”

Section: Discussionsupporting

confidence: 83%

Hiperplasia angiolinfoide com eosinofilia: um caso raro em cavidade oral

et al. 2016

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Angiolymphoid Hyperplasia with Eosinophilia

Alshaiji¹

2019

Harper's Textbook of Pediatric Dermatology

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Epithelioid haemangioma (angiolymphoid hyperplasia with eosinophilia subtype) involving the vermillion border of the lower lip

2021

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Epithelioid vascular tumours include benign epithelioid haemangioma (EH) and epithelioid angiomatous nodule, low-grade malignant epithelioid haemangioendothelioma and high-grade epithelioid angiosarcoma. These tumours share epithelioid cell morphologies and intracytoplasmic vacuoles, with distinction based on differing architectural and nuclear features. This report highlights a case of EH, angiolymphoid hyperplasia with eosinophilia subtype, involving the vermillion border of the lower lip in an elderly female patient. The clinical and histological features, differential diagnosis, aetiopathogenesis, molecular basis and treatment options are discussed as part of a comprehensive review of this rare entity.

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Angiolymphoid hyperplasia with eosinophilia in the angle region of the mandible

Cited by 5 publications

References 14 publications

Hiperplasia angiolinfoide com eosinofilia: um caso raro em cavidade oral

Hiperplasia angiolinfoide com eosinofilia: um caso raro em cavidade oral

Angiolymphoid Hyperplasia with Eosinophilia

Epithelioid haemangioma (angiolymphoid hyperplasia with eosinophilia subtype) involving the vermillion border of the lower lip

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