This case report describes a rare presentation of angiolymphoid hyperplasia with eosinophilia arising from a port wine stain, associated with elevated serum renin levels that normalized after excision.
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May explain symptomatic thickening in port wine stains; extends sparse reports but leaves open renin association.
A 19-year-old male with a port wine stain on the base of his neck presented with a 5-month history of gradual thickening of the involved skin which interfered with clothing and caused repeated bleeding. The lesion was excised and histopathologic examination revealed angiolymphoid hyperplasia with eosinophilia (ALHE) arising from the pre-existing port wine stain - a rare finding with only one previously reported case. Additionally the lesion was associated with elevated serum renin levels which virtually normalized following excision of the lesion. We further demonstrated the expression of angiotensin converting enzyme and angiotensin II receptors 1 and 2 by the lesion and discuss the possible role of the renin-angiotensin system in this condition.
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Manton et al. (2015) studied this question.
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