A swollen swimmer

Abstract

In November, 1997, a 38-year-old former UK swimming international presented with a history of intermittent angioedema involving his face, neck, and limbs. This was associated with wheezing. A diagnosis of late-onset asthma had already been made. His treatment had been daily bronchodilators and up to seven courses of oral corticosteroids per year for the preceding 5 years. In addition, at the age of 27 he had had an unexplained “autoimmune illness”, for which he had been treated with corticosteroids at another hospital. Physical examination was normal with the exception of a forced expiratory wheeze. No cause could be found for his angioedema in his medication, specific allergic stimulus, or defect or deficiency in C1-inhibitor activity. He had eosinophilia (eosinophils 2·4 10/L, 19% of total white-cell count). He was treated with tranexamic acid and prophylactic antihistamines with substantial improvement. Corticosteroids were withdrawn. 6 months later his angioedema recurred, affecting sites of minor trauma and occurring after sexual activity. He also complained of malaise, and examination showed cardiomegaly and muffled heart sounds. A moderate pericardial effusion with severe left-ventricular dysfunction was confirmed by echocardiography. He was admitted for investigation. On further questioning he denied any history of alcohol or drug abuse, and specifically denied use of anabolic steroids or leukotriene antagonists. Peripheral-blood eosinophilia was again found. While in hospital he had an episode of severe bronchospasm, associated with moderate hypoxaemia, and transient patchy pulmonary infiltrates on his chest radiograph. He responded to treatment with corticosteroids, aminophylline, and bronchodilators.