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Angiolymphoid hyperplasia with eosinophilia in pregnancy

To the Editor Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon benign disorder of young adults with female predominance, characterized by solitary or multiple nodules on the head and neck region. Some cases associated with pregnancy have been reported earlier.1 During the first semester of the second pregnancy, the 28year-old female patient developed two subcutaneous, livid nodules 1–1.5 cm in diameter at the lateral side of the left eyebrow and in the left temporal region, as well as several smaller lesions on the scalp of the left parietal region. Sometimes they were spontaneously bleeding. The overlying skin was purplish or normal, larger lesions were pulsating (fig. 1). The two largest nodules were surgically removed. The histological picture showed circumscribed, lobuled lesions, oedematous endothelial cells in the dermal blood vessels and lymphocytic and eosinophilic infiltration (fig. 2). Routine laboratory investigations, peripheral eosinophil count revealed negative results. Direct immunofluorescent histology did not show overexpression of oestrogen or progesterone receptors in the lesions. Polymerase chain reaction (PCR) investigations of human herpesvirus 8 (HHV8) and human T cell lymphoma virus (HTLV) were also negative. After the patient has delivered, the lesions showed spontaneous regression. ALHE was originally described by Wells and Whimster in 1969.2 This rare benign tumour occurs with a female predominance, and most often affects the head and neck region. The typical clinical signs are solitary or multiple purplish, brownish papules and subcutaneous nodules. The histological picture is characterized by the proliferation of endothelial cells associated with lymphocytic and eosinophilic infiltrate. Results of immunohistological examinations, showed that perivascular infiltrating lymphocytes are mostly CD4 + positive. Proliferating endothel cells express adhesion molecules: VLA-1, -3, -5, ICAM-1, ELAM-1.3 Sometimes marked peripheral eosinophilia can be detected. Kimura’s disease was earlier thought to be related to ALHE, but nowadays it is considered a different clinical entity. The main differences between the two diseases were described by Chum et al. in 1992.4,5 Some earlier clinical entities with other names are identified as ALHE, some authors suggested the nomenclature of epithelioid haemangioma.6 The origin of the disease is not known, infection (HTLV or HHV8), hormonal background, or possible role of trauma are the mostly discussed possibilities. Increased expression of vascular endothelial growth factor (VEGF) and interleukin 5 (IL-5) was detected in some cases.7 Associated cases of the disease to pregnancy were already described. Moy et al. found oestrogen and progesterone receptor overexpression in the lesional skin of pregnant women.1 In our case, direct immunofluorescent histology did not show overexpression of oestrogen or progesterone receptors in the lesions. HHV8 and HTLV PCR investigations were also negative. The possible treatment modalities were intralesional steroid or IFN α, surgical excision, cryotherapy and laser therapy.8 Association of ALHE with nephrotic syndrome has also been described.9 The interest of our case makes the typical clinical picture and outcome of the rare disease. Its association with pregnancy suggests the pathogenetic role of immunological and endocrinological changes in the background.