Angiolymphoid hyperplasia with eosinophilia occurring on the penis

ARTICLE

Auteur(s) : Shun Ohmori, Kazunari Sugita, Yu Sawada, Ryosuke Hino, Motonobu Nakamura, Yoshiki Tokura

Department of Dermatology, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu 807-8555, Japan

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign lymphoid and vascular lesion that was first described in 1969 by Wells and Whimster. Histologically, ALHE is characterized by the proliferation of vascular endothelial cells protruding into the lumen and the massive infiltration of lymphocytes and eosinophils, occasionally with lymphoid follicles. Blood eosinophilia is seen in approximately 20% of cases, but the levels of serum immunoglobulin E (IgE) are usually normal. ALHE presents with small erythematous dermal papules or nodules, and arises mostly on the head and neck, and exceptionally on the trunk and extremities [1]. Therefore, the predilection sites are a hallmark for making the diagnosis, and dermatologists may miss a differential diagnosis of ALHE for lesions occurring on the other sites. Here, we report a case of ALHE affecting the penis. Our literature review shows that the penis is one of the notable sites of ALHE.

A 20-year-old Japanese man was referred to us with a 6-month history of a rapidly growing nodule on the penis. He had been initially diagnosed as having scabies and had taken oral ivermectin twice in a monthly interval without any therapeutic effect. The patient had no history of a sexually transmitted disease or trauma caused by harsh sexual intercourse. Clinical examination revealed a soft erythematous nodule, 1.6 × 1.4 cm in diameter, on his penile shaft (figure 1A). He had no lymphadenopathy. Laboratory data, including blood cell counts and chemistry, were normal, except for a high serum IgE level (853 kU/L; normal, 0-170 kU/L). Serological tests for syphilis were negative.

An excisional biopsy of the lesion showed dense infiltration of lymphocytes and eosinophils (figure 1B), and proliferation of small vessels with swollen endothelial cells protruding into the lumen (figure 1C). Immunohistochemical studies revealed that lymphoid follicles composed of CD20⁺ B cells (figure 1D) were surrounded by numerous CD4⁺ T cells. On the basis of these histological findings, we diagnosed the lesion as ALHE. Immunostaining with anti-CCL11 antibodies (Santa Cruz Biotechnology, Santa Cruz, CA) revealed an infiltrate of eotaxin/CCL11-expressing cells (figure 1E). This suggests that not only CCL11 but also CCL11-chemoattracted eosinophils may induce a local angiogenetic response and the resultant tumor formation [2].

Our review of literature found five cases of ALHE arising on the penis (table 1) [1, 3-6]. The mean age at diagnosis was 27 years, ranging from 9 to 47 years, which is not different form that of ALHE on the head and neck. Blood eosinophilia was observed only in case 1. Serum IgE levels were not described in the reported cases. Both the shaft and glans are the affected sites, and the size ranged from 1 to 1.6 cm diameter. Only local excision gave a satisfactory improvement in three patients, while radiotherapy or topical steroids showed little effect. Although the penis is an unexpected site for ALHE, this disease should be included in the differential diagnosis of a reddish nodule of the penis, especially when the lesion is resistant to conventional therapies.
Table 1 Reported cases of ALHE occurring on the penis

Acknowledgements

References

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