Abstract
A 40-year-old gentleman presented with a history of multiple swellings involving his face, scalp, left axilla, back, and right thigh for the past 8 years. For the last 6 months, he developed intermittent low-grade fever, anorexia, weight loss, and gradually worsening breathlessness. On evaluation, the patient was found to have abnormally elevated absolute eosinophil count. Workup for the etiology of eosinophilia was unrewarding. All investigations related to an underlying myeloproliferative disorder were negative. Hence, a clinical possibility of angiolymphoid hyperplasia with eosinophilia (ALHE) was kept which was confirmed on histopathology. In the absence of other causes of hypereosinophilia, a clinical diagnosis of "associated hypereosinophilic syndrome" secondary to ALHE was made. He was managed with oral corticosteroids. The absolute eosinophil count reduced markedly, while the swellings showed a more gradual response, shrinking in size by about 50% following two months of therapy. The index case thus highlights a rather unusual presentation of ALHE.