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Dear Editor, Angiolymphoid hyperplasia with eosinophilia (ALHE) is an unusual vascular proliferation, developing predominantly in the dermis and subcutaneous tissues of the periauricular area and the scalp. Histologically, ALHE is characterized by plump endothelial cells protruding into the lumen of vessels and dense infiltration of lymphocytes and eosinophils in the stroma. Here, to the best of our knowledge, we report the first case of intravascular ALHE in both periauricular regions. A 60-year-old man presented with a 2-year history of asymptomatic multiple subcutaneous nodules on the scalp and both periauricular regions (Fig. 1a,b) and a history of untreated hypertension experienced for several years. He had eosinophilia (3.4 9 10 3 /µL, 34% of the white blood cell count) and high serum immunoglobulin E levels (2200 mg/dL). Magnetic resonance imaging revealed multiple subcutaneous nodules, which had many lumens inside (Fig. 1c,d). There was no regional lymphadenopathy. Magnetic resonance angiography showed marked tortuous subcutaneous arteries (Fig. 1e) of the scalp, which were the branches of the external carotid arteries. Arteriovenous shunts or aneurysms were not found in the cerebral arteries. The biopsy specimen of a subcutaneous mass of the right periauricular lesion showed a nodule containing small vessels and inflammatory cell infiltration (Fig. 1f,g). A high-power view revealed that the small vessels were lined by plump endothelial cells with dense infiltration of eosinophils and lymphocytes in the stroma (Fig. 1h, i). Smooth muscle actin (SMA) staining confirmed that the nodule itself was an artery with a thick muscular wall. The muscular artery became dilated and was almost completely occluded with SMA-positive small proliferating vessels (Fig. 1j). The muscular artery included some small arteries. No lymphoid follicle formation was found in the specimen. Accordingly, we diagnosed the lesion as intravascular ALHE.

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