ALHE in a breast: A first case report in a childhood

Abstract

A 3‐year‐old boy presented with on and off fever, anorexia, and diffuse left breast swelling for 3 months. Overlying skin was shiny and red (Figure 1). On palpation, swelling was cystic to firm and slightly mobile with well‐defined borders. There was no regional lymphadenopathy. Systemic examination was within normal limit. Hematological investigation showed moderate anemia (hemoglobin‐9.0 g/ dL), total leukocyte count of 14 500/cmm with 19% eosinophils (AEC was 2755/cumm), and peripheral smear revealed microcytic hypochromic blood picture. All other laboratory parameters were within normal limit. FNAC from the left breast swelling showed mixed inflammatory infiltrate comprising of plasma cells, macrophages, giant cells, numerous eosinophils, and ill‐formed granulomas. However, no parasite was seen and stain for acid fast bacilli (AFB) was negative. Ultrasonography suggested lesion to be of inflammatory origin. Patient underwent wide local excision of the mass. Histopathological examination revealed vaguely circumscribed, unencapsulated lesion with proliferating vascular channels lined by plump endothelial cells (Figure 2A). Intervening areas showed dense inflammatory infiltrate in the dermis and subcutis of breast composed of lymphocytes, plasma cells, histiocytes, polymorphs, and numerous eosinophils (Figure 2B) along with many noncaseating epithelioid cell granuloma with foreign body giant cell reaction (Figure 2C). Periductal lymphoid infiltrate and lymphoid aggregates without germinal center formation was also noted (Figure 2D). No parasite or fungus was seen. Stain for AFB was negative. Based on the characterstic histomorphological findings, diagnosis of Angiolymphoid hyperplasia with eosinophilia (ALHE) was rendered. The child parents were counseled regarding the benign nature of disease. The patient is doing well at 4 months of follow‐up, without any recurrence. Angiolymphoid hyperplasia with eosinophilia is a rare, benign vasoproliferative disorder that usually occurs in 3rd to 5th decades of life with a female preponderance. Clinically it presents as single or multiple, pinkish papules or nodules in the head and neck region. Other rare sites reported include the orbit, oral mucosa, hands, liver, spleen, heart, bone, and breast. To the best of our knowledge only two case reports of ALHE in adult female breast has been described in literature so far. However, childhood ALHE has not been reported till date. Our case also showed granulomatous reaction and many giant cells, which is rarely reported in previous case reports. The pathogenesis of ALHE has been variously attributed to prior trauma, infectious agents, atopy, reactive hyperplasia, and benign vascular neoplasia. Recently it was postulated to be a low‐grade T cell lymphoproliferative disorder. Majority of cases present as slow growing persistent mass lesion and sometimes it may regress spontaneously. Complete local excision and follow up is the most optimal management. However, recurrence rate is high probably due to difficulty in identifying the margins of this highly vascular lesion. Recently pulsed dye and other lasers have been shown effective treatment options.