儿童变应性支气管肺曲霉菌病四例报告并文献复习

Allergic bronchopulmonary aspergillosis：a report of four cases with literature review

目的 分析儿童变应性支气管肺曲霉菌病（ABPA）的临床特点.方法 回顾性分析首都医科大学附属北京儿童医院呼吸二科病房2010年4月至2014年3月确诊的4例ABPA患儿的临床资料.分析近10年国内外数据库中报道的儿童ABPA检索资料.结果（1）4例ABPA患儿,2例确诊为囊性纤维化,1例同时诊断为哮喘.临床表现：咳嗽3例,胸闷（黏液栓堵塞）1例,反复喘息史2例;4例肺部影像学均表现有中心性支气管扩张,其中1例表现为游走性片状阴影;4例患儿血清总IgE升高,烟曲霉抗原速发皮试、血烟曲霉特异性IgE和IgG均为阳性.4例均使用糖皮质激素及抗真菌药物（伊曲康唑或伏立康唑）口服治疗,病情均得到不同程度的缓解.（2）在近10年国内外数据库中检索儿童ABPA,国外病例报告15篇,共报道22例,其中16例发生于囊性纤维化,3例发生于哮喘,16例囊性纤维化合并ABPA患儿中,仅1例以ABPA为首发症状.国内ABPA病例报告仅1例.结论 对于哮喘、过敏性疾病的患儿,如伴有IgE的异常增高、影像学出现支气管扩张、反复肺不张时需考虑ABPA的可能;对于临床确诊为ABPA的患儿,应注意除外囊性纤维化.

Objective To understand the clinical characteristics of allergic bronchopulmonary aspergillosis（ABPA） so as to diagnose and treat the disease earlier.Method A retrospective study was conducted on ABPA patients diagnosed in the Second Department of Respiratory Medicine,Beijing Children＇s Hospital Affiliated to Capital Medical University from April 2010 to March 2014.The literature of children＇s ABPA retrieved from the databases at home and abroad in recent 10 years were analyzed.Result （1）Among the 4 cases of ABPA,cystic fibrosis （CF） and asthma were diagnosed in 2 and 1 cases,respectively.Cough was present in 3 patients,recurrent wheezing in 2 and chest tightness in 1 case.CT scans showed central bronchiectasis in all 4 cases,while 1 patient had migratory shadows.All cases had elevated serum total IgE,immediate cutaneous reaction to aspergillus fumigatus;A.fumigatus-specific IgE and IgG were positive in 4 cases.The diagnosis of the 4 cases was confirmed according to the history,radiologic investigations and laboratory findings.All of them were improved after the treatment with glucocorticosteroid and antifungal agents （voriconazole or itraconazole）.（2） We retrieved articles on the ABPA in the databases at home and abroad published in the recent 10 years,there were 22 foreign reports and only one case in domestic report.Among the 22 foreign cases,16 patients were CF,3 were asthmatics.ABPA was diagnosed as the initial presentation in only one case with CF.Conclusion In asthmatics or the patients with allergic disease,if there are highly elevated serum total IgE,central bronchiectasis or recurrent atelectasis in chest imaging,the patients should be further investigated for ABPA.The diagnosed cases of ABPA should be screened for CF routinely.