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2019 Month : December Volume : 8 Issue : 52 Page : 4020-4022

Angiolymphoid Hyperplasia with Eosinophilia Treated with Isotretinoin.

Sunil N. Petkar1, Venkatesh Giri2, Pranit Farande3, Jigar Patel4, Asha Kushwaha5

Corresponding Author:
Dr. Pranit Farande,
Shankarsheela Complex, Shop No. 1,
Sector-8, Airoli, Navi Mumbai-400708,
Maharashtra, India.


Angiolymphoid Hyperplasia with Eosinophilia (ALHE) is an uncommon benign clinical entity characterized by the presence of a variable number of papules, plaques or nodules of the dermis and subcutaneous tissues.[1] ALHE shows a predilection for the head and neck area.[2] ALHE is a benign neoplasm of the vascular endothelium. ALHE is also called as epithelioid hemangioma, a term that better describes the most distinguishing feature of this entity -the abnormal proliferation of endothelial cells.[3] The etiology of ALHE is obscure; however, it has been reported to occur in pregnancy and also following trauma in some cases.[2]

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare and idiopathic vascular disorder. It is characterized by red to brown papules and nodules, typically localized on the head and neck, particularly around the ear as singular or multiple lesions. Although ALHE is a benign disease, lesions are often persistent and difficult to eradicate. Young to middle age women are more commonly affected. The histological examination corresponds to a florid vascular proliferation with atypical endothelial cells surrounded by a lymphocytic and eosinophilic infiltrate. We describe a young male with solitary nodular lesion over the left side of neck below left ear with scar mimicking scrofuloderma; the histological examination was consistent with ALHE.

                ALHE was first described by Wells and Whimster[4] who defined it as a late stage of Kimura disease. Since then there has been a lot of controversy about these two conditions. Nowadays it is widely accepted to be different entities. ALHE is a rare, benign, vasoproliferative disease of unknown origin. It is characterized by intradermal or subcutaneous single or multiple reddish-brown papules or nodules, mainly on the head and neck, especially around the ears.[5,6] Less often lesions of ALHE have been described in the mouth, on the trunk, extremities, vulva and penis.

Affects mostly young to middle-aged females of any race, may complain about itching, tenderness or pulsation of the lesions. They do not have lymphadenopathy or peripheral eosinophilia in comparison with Kimura disease.[5,6,7]

The pathogenetic mechanism of ALHE is unknown. In some cases, there is a preceding trauma, a recently published article described ALHE after an ear piercing.[8] Some reporters suggest it is related to infection and high estrogen status. Nevertheless, there seems to be consensus that ALHE is a reactive rather than a neoplastic process.[6,9,10]

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