• Korean Journal of Head & Neck Oncology
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• v.32 no.2
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• pp.47-50
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• 2016

Angiokeratomas represent vascular lesions and are histologically characterized by superficial dermal vascular ectasia with overlying epidermal hyperkeratosis. Angiokeratoma circumscriptum is one of five types in the group of the angiokeratomas. The lesion is present from birth but may appear in childhood or adulthood. Angiokeratoma circumscriptum is typically hyperkeratotic papules or nodules situated unilaterally on the foot or distal aspect of the leg, but can occur elsewhere on the body. Isolated tongue involvement is very rare. We report a 12-year-old boy with angiokeratoma circumscriptum of the posterior dorsal tongue with a review of the literatures.

• Journal of the Korean Association of Oral and Maxillofacial Surgeons
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• v.40 no.5
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• pp.240-245
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• 2014

Angiokeratoma is a benign cutaneous lesion of the capillaries, presenting as dilated vessels in the upper part of the dermis. Although this disorder is classified into various types and has been occasionally reported in the skin of the scrotum or extremities, the involvement of the oral cavity mucosa has been rarely reported. The present study reports a case of angiokeratoma circumscriptum in the buccal mucosa. The expression of vascular endothelial growth factor (VEGF) and both of its receptors (VEGFR-1 and VEGFR-2) was demonstrated by immunohistochemistry in the endothelial cells lining the dilated vessels. The expression of VEGFR-2 was higher than that of VEGFR-1 in the endothelial cells in the lesion, indicating an increased rate of endothelial cell proliferation within the lesion. Interestingly, some of the endothelial cells co-expressed VEGF and its two receptors. These results suggest that endothelial cells in the pathologically dilated vessels possess VEGF autocrine growth activity involved in vasculogenesis and maintenance in angiokeratoma lesions. To our knowledge, this is the second report published on isolated oral angiokeratoma confined to the buccal mucosa and the first case report on angiokeratoma circumscriptum involving the buccal mucosa.