儿童滤泡性细支气管炎2例并文献复习

Follicular Bronchiolitis: Report of 2 Pediatric Cases and Literature Review

目的总结儿童滤泡性细支气管炎(FB)的临床表现、影像学表现、肺功能、治疗及预后情况,提高儿科医师对该病的认识。方法回顾性分析上海儿童医学中心2016年经胸腔镜肺活检确诊的2例儿童FB患者的临床资料,并以"滤泡性细支气管炎"为检索词在中国知网数据库,以"Follicular bronchiolitis"为检索词在Pubmed数据库中检索1974年1月1日至2017年12月31日的相关文献,结合文献总结其疾病特点。结果 2例女性患者,年龄分别为14岁、12岁,病程均为3年,表现为间断发热、咳嗽伴脓痰,胸部高分辨率CT均表现为多发小结节影伴实变、支气管扩张及小气道改变,肺活检病理示淋巴组织增生相关疾病,细支气管周围淋巴滤泡形成,2例均给予小剂量红霉素维持治疗,随访1年CT好转不明显。检索资料较完整的儿童FB文献共15篇35例,主要表现为咳嗽、气促、喘息、呼吸困难、反复呼吸道感染、反复发热及生长发育受限。影像学表现常见双肺弥漫性小结节、支气管扩张、网状影及磨玻璃影等。可使用激素、抗生素、免疫抑制剂、支气管扩张剂等治疗,多数患者病程长,但总体预后较好。结论 FB为儿童少见的小气道疾病,临床表现及肺功能无特异性,影像学以小结节、支气管扩张及间质改变多见,需要病理诊断,目前无特效治疗手段,但总体预后较好。

Objective To summarize the clinical manifestations,imaging findings,pulmonary function,treatment and prognosis of follicular bronchiolitis（FB） in children,and improve the understanding of the disease by pediatricians.Methods In this report,we presented the clinical data and findings of two girls with FB confirmed by thoracoscopic lung biopsy in Shanghai Children＇s Medical Center in 2016,and a literature search was performed with ＂ follicular bronchiolitis＂ as the key words in China Knowledge Resource Integrated Database and Pubmed. The time interval was from January 1 st1974 to December 31 st 2017 to summarize the features of the disease. Results The two girls,aging 14 years and 12 years,both presented with productive cough and fever with a disease course of 3 years. The findings of high resolution CT showed multiple small nodules consolidation,bronchiectasis and small airway changes. The pathological examination revealed peribronchiolar lymphoid follicles. 2 cases were treated with a small dose of erythromycin. After 1 year of follow-up,CT improvement was not obvious. 15 studies of 35 pediatric FB cases were included in the literature review. The main clinical manifestations were cough,shortness of breath,wheezing,dyspnea,recurrent respiratory infection,recurrent fever and growth restriction. The typical imaging findings were multiple small nodules,bronchiectasis,reticular opacities,and ground glass opacities. The medical treatments included corticosteroids,antibiotics,immunosuppressive agents,bronchodilators,etc. Most FB patients had long clinical course of disease,but the overall prognosis was good. Conclusion FB is a rare small airway disease in children. There is no specificity in clinical manifestations and lung function. High resolution CT findings are mainly small nodules,bronchiectasis and interstitial changes. Histopathological examination is necessary. There is no specific effective treatment at present,but the overall prognosis is good.