Angiolymphoid hyperplasia with eosinphilia presenting with postauricular swelling

Angiolymphoid hyperplasia with eosinophilia (ALHE) is an unusual vascular proliferative condition. The entity is almost unknown to the Otolaryngologist though it has a predilection for the head and neck particularly the region around the ear. In this case report ALHE presented as a postauricular swelling. A 48-year-old Saudi male was referred from General Surgery to the ENT clinic with a painless swelling over the right postauricular region of 2 months duration. The swelling was gradually increasing in size and was associated with itching occasionally. On physical examination there was a superficial swelling on the right mastoid region, 3 x 2 cms in size. The skin over the swelling was free and unremarkable. It was nontender, mobile, firm and rubbery in consistency, with well circumscribed margins. No other swellings were noted in the head and neck region. Ear, nose and throat and systemic examination was normal. Patient was diabetic (non insulin dependant diabetes mellitus). Routine blood test was carried out. Fine needle aspiration cytology (FNAC) was suggestive of a benign mesenchymal lesion. As FNAC was not conclusive and a 7 day course of antibiotics and anti-inflammatory drugs showed no response, we opted for excisional biopsy under local anesthesia. A well-circumscribed subcutaneous mass was found: the operative field was unduly bloody, there were 2 big vessels supplying the mass which were ligated and small bleeders were cauterized. The mass was completely excised, and the specimen was sent for histopathology. The mass measured 2 x 1.5 x 1 cm and was firm. On section it was grey white with a tan periphery. The histology was characterized by an exuberant proliferation of small blood vessels, and a heavy chronic inflammatory cell infiltrate. Many of the vessels were lined by plump endothelial cells projecting into the lumen like tombstones the cells had an epithelioid appearance with abundant pale eosinophilic cytoplasm and large round vesicular nuclei. The infiltrate consisted of lymphocytes, plasma cells and eosinophils, the last were particularly numerous (Figure 1). The diagnosis was epithelioid hemangioma (ALHE). There was no evidence of arteriovenous malformation. Subsequent follow up for 6 months was uneventful. Angiolymphoid hyperplasia with eosinphilia is an unusual condition poorly recognized by the Otolaryngologists. The term was first used in 1969 by Clinical Notes