Longley S, Panush R S
Ann Allergy. 1987 Apr;58(4):257-9.
We identified two siblings with exercise-induced anaphylaxis who share the HLA haplotype A3-B8-DR3 with their atopic father. The index case, a 16-year-old female, noted initial episodes at age 13. Intense pruritus, urticaria, facial edema, choking sensation, nausea, hypothermia, and collapse followed vigorous running but not swimming, cycling, racquetball, solar exposure, or cold exposure. Neither antihistamine, antiserotonin, anticholinergic nor epinephrine therapy was entirely effective or protective; only modification of running prevented episodes. Three similar episodes were noted at age 15 years by a brother who, now age 25, relates a 4-year history of seasonal rhinitis and exercise-related urticaria without anaphylactoid reaction. The remainder of the family (father, 47; mother, 46; brother, 22 years) does not have exercise intolerance. The father has allergic rhinitis; his nephew suffers exercise-induced urticaria without collapse. HLA typing revealed the father to be A1-B8-DR3, A3-B8-DR3; the symptomatic daughter to be A3-B8-DR3, A30-B5-DR8; and the symptomatic son to be A3-B8-DR3, A30-B5-DR8. The asymptomatic mother was A30-B5-DR8, A2-B7-DR5 and the asymptomatic son A1-B8-DR3, A30-B5-DR8. We describe exercise-induced anaphylaxis in a unique familial setting, perhaps linked to the HLA haplotype A3-B8-DR3.
我们确定了两名患有运动诱发性过敏反应的兄弟姐妹,他们与患有特应性疾病的父亲共享HLA单倍型A3 - B8 - DR3。索引病例是一名16岁女性,13岁时首次发病。剧烈瘙痒、荨麻疹、面部水肿、哽咽感、恶心、体温过低和虚脱在剧烈跑步后出现,但游泳、骑自行车、壁球、日晒或冷暴露后未出现。抗组胺药、抗血清素药、抗胆碱能药和肾上腺素治疗均未完全有效或起到保护作用;只有改变跑步方式才能预防发作。一名哥哥在15岁时也出现了三次类似发作,他现在25岁,有4年季节性鼻炎和运动相关性荨麻疹病史,但无类过敏反应。家族中的其他成员(父亲,47岁;母亲,46岁;哥哥,22岁)没有运动不耐受情况。父亲患有过敏性鼻炎;他的侄子患有运动诱发性荨麻疹但无虚脱症状。HLA分型显示父亲为A1 - B8 - DR3,A3 - B8 - DR3;有症状的女儿为A3 - B8 - DR3,A30 - B5 - DR8;有症状的儿子为A3 - B8 - DR3,A30 - B5 - DR8。无症状的母亲为A30 - B5 - DR8,A2 - B7 - DR5,无症状的儿子为A1 - B8 - DR3,A30 - B5 - DR8。我们描述了一种独特的家族性运动诱发性过敏反应,可能与HLA单倍型A3 - B8 - DR3有关。