A swollen swimmer

A swollen swimmer
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浮肿的游泳者

DOI:
10.1016/s0140-6736(99)02164-9
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发表时间:
1999
期刊:
The Lancet
影响因子:
--
通讯作者:
S. Marshall
S. Marshall
中科院分区:
--
文献类型:
--
作者:
Jeffrey L. Greenwald;Nicky West;A. Bird;S. Marshall

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1997年11月,一名38岁的前英国游泳国际选手因面部、颈部和四肢间歇性血管性水肿病史就诊。这与喘息有关。已经诊断出迟发性哮喘。他的治疗是每日支气管扩张剂和多达7个疗程的口服皮质类固醇,每年为前5年。此外,他在27岁时患有无法解释的“自身免疫性疾病”,为此他在另一家医院接受了皮质类固醇治疗。除用力呼气性喘鸣外,体格检查正常。在他的药物、特定过敏刺激或C1抑制剂活性缺陷或缺乏中未发现血管性水肿的原因。他患有嗜酸性粒细胞增多症(嗜酸性粒细胞2· 4109/L,占总白细胞计数的19%)。患者接受氨甲环酸和预防性抗组胺药治疗,病情得到实质性改善。停用皮质类固醇。6个月后,他的血管性水肿复发,影响轻微创伤的部位,发生在性活动后。他还抱怨不适,检查显示心脏肥大和心音低沉。超声心动图证实中度心包积液伴严重左心室功能障碍。他被允许接受调查。在进一步询问时,他否认有任何酗酒或吸毒史,并特别否认使用合成代谢类固醇或白三烯拮抗剂。再次发现外周血嗜酸性粒细胞增多症,住院期间,患者出现严重支气管痉挛,伴有中度低氧血症,胸片显示一过性斑片状肺浸润。他对皮质类固醇、氨茶碱和支气管扩张剂的治疗有反应,并获得了他11年前的住院记录。这些记录了全身性疾病,包括肌痛、发热、腹泻、嗅觉丧失和间歇性大腿感觉过敏,伴有双侧网状结节性肺浸润和一过性心包积液。经支气管肺活检与嗜酸性粒细胞性肺炎一致,显示血管外嗜酸性粒细胞,但无肉芽肿。诊断为非特异性自身免疫性血管炎,并给予静脉和口服皮质类固醇治疗。他的症状完全消失,尽管他没有参加随访,但7年后用于评估哮喘的胸部X光片显示心脏大小正常。在随后的录取中,他
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· 4109/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 Corticosteroids were withdrawn. 6 months later his angioedema recurred, affecting sites of minor trauma and occurring after sexual activity.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.The records of his admission to hospital 11 years previously were obtained. These recorded a systemic illness with myalgia, fever, diarrhoea, anosmia, and intermittent hyperaesthesia of his thigh associated with bilateral reticulonodular pulmonary infiltrates and a transient pericardial effusion. A transbronchial lung biopsy was consistent with an eosinophilic pneumonia, showing extravascular eosinophils but no granulomas. A diagnosis of non-specific autoimmune vasculitis had been made, and he was treated with intravenous and oral corticosteroids. His symptoms resolved completely, and although he did not attend for follow-up, a chest radiograph 7 years later for evaluation of asthma showed a normal heart size. During his subsequent admission, his
DOI: 10.1056/nejm198406213102501
发表时间: 1984-01-01
影响因子: 158.5
作者:
GLEICH, GJ;SCHROETER, AL;KOHLER, PF
通讯作者: KOHLER, PF