Hiperplasia angiolinfoide com eosinofilia: um caso raro em cavidade oral

Abstract

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.

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

“…Since then EH has been reported under a plethora of terms, including ALHE, atypical or pseudopyogenic granuloma, inflammatory angiomatous nodule, popular angioplasia, inflammatory arteriovenous haemangioma and intravenous atypical vascular proliferation. 4,12,13,15 In many instances EH has been grouped with histologically similar lesions such as Kimura disease, under the umbrella term 'ALHE'. 4,8,15 However, currently, most studies agree that they are distinct diseases with different epidemiological, clinical and histopathological changes.…”

“…Epithelioid haemangioma most commonly presents in mid-adult life as single or multiple erythematous/ hyperpigmented dome-shaped papules or nodules with an average size of 1 cm. 3,4,7,12,13,16,17 When multiple, they tend to be grouped, often coalescing, raising concern for a malignant process. 3,15,18 EH frequently involves the head and neck region, penis and the extremities, especially the arms and hands.…”

“…In the head and neck region, any site may be affected including the maxillary bones, salivary glands, muscles and skin. 9,13 Oral lesions are rare, with only 28 cases of oral EH described in literature. 19 The most common oral sites include the lips, followed by the buccal mucosa and tongue.…”

“…19 The most common oral sites include the lips, followed by the buccal mucosa and tongue. 12,13 Patients present over a wide range of ages, with a peak in the third to fifth decades of life. 4,9,10,17 Several reports indicate a predilection for women, although some have reported a male preponderance.…”

“…[2][3][4][5][6][7][8][9][10][11] Many other terms have been used in literature to describe this lesion, emphasising the confusion that exists regarding this unusual condition. 4,12,13 Distinguishing EH from other malignant epithelioid vascular tumours is paramount due to differences in their management and clinical outcome. 2 This report highlights a case of EH, angiolymphoid hyperplasia with eosinophilia (ALHE) subtype, involving the vermillion border of the lower lip in an elderly female patient.…”