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Angiolymphoid hyperplasia with eosinophilia successfully treated with tacrolimus ointment

Editor Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare, benign, vascular, proliferative disease of unknown aetiology. It commonly occurs among women in their 20s to 40s, presenting as single or multiple pink to reddish-brown papules or nodules mainly on the head and neck region, with particular predilection for the peri-auricular area. The lesions are often pruritic or painful with little propensity for spontaneous resolution. Treatment for ALHE is known to be difficult because of frequent recurrences. Here we report a Chinese man with ALHE who was successfully treated with topical application of 0.1% tacrolimus ointment. A 22-year-old Chinese man was referred to our hospital with a 2-year history of enlarging reddish, itchy papules and nodules on his left ear and post-auricular area. The lesions were painless and some of them were occasionally bleeding. Skin examination demonstrated multiple superficial erythematous papules and nodules on his left ear and post-auricular region (Fig. 1). There was no regional lymphadenopathy. His family history was not significant. Histology from lesional skin showed vascular proliferation and a mixed dermal inflammatory infiltrate consisting primarily of lymphocytes and eosinophils. The blood vessels lined by plump endothelial cells, which protruded into the lumina and resulted in a characteristic ‘tombstone’ appearance. Laboratory tests showed total white blood cell count of 5.45 · 10/L with eosinophilia (7.9%). Serum immunoglobulin E (IgE) was 2066 kU/L (normal < 150). All other standard blood, serum and clotting parameters were within normal limits. A diagnosis of ALHE was made on clinical and histological features. The patient started treatment with 0.1% tacrolimus ointment twice daily. The pruritic sensation was dramatically relieved within 2 weeks. After 14 weeks of topical tacrolimus use, papules and nodules had basically disappeared (Fig. 2). Treatment with tacrolimus was discontinued 10 months later. There have been no adverse effects or clinical evidence of recurrence during the 15-month follow-up period. ALHE is a benign, vascular, proliferative disease associated with interstitial inflammation of lymphocytes and eosinophils. In addition to tissue eosinophilia, some patients may develop peripheral blood eosinophilia and markedly elevated serum IgE levels. Although a number of proposed treatment modalities have been used, therapy for ALHE is still challenging. Surgical excision is considered the most effective treatment option, but a recurrence rate of 50% was reported. Variable effects are achieved with other methods, such as cryotherapy, radiotherapy, pulse dye laser, carbon dioxide laser, retinoids, glucocorticoids (topical or intralesional), interferon-a, anti-interleukin-5 (IL-5) antibody and imiquimod. Topical tacrolimus has proved to be an effective and safe treatment option for atopic dermatitis. Its significant antiinflammatory activity and immunomodulatory capabilities make it an ideal treatment modality for a series of inflammatory and Figure 1 Multiple erythematous papules and nodules on the left ear. Figure 2 Marked improvement after treatment with tacrolimus ointment.