儿童先天性气管狭窄合并交通性支气管肺前肠畸形2例并文献复习  

Congenital tracheal stenosis complicated with communicating bronchopulmonary foregut malformation in 2 children and literature review

作　　者：王超[1] 冯致余[1] 翟允鹏[1] 杨彦亮 王少超[1] 王磊[1] 孟晨[1] 张士松[1] Wang Chao;Feng Zhiyu;Zhai Yunpeng;Yang Yanliang;Wang Shaochao;Wang Lei;Meng Chen;Zhang Shisong(Center for Respiratory Intervention,Children′s Hospital Affiliated to Shandong University(Ji′nan Children′s Hospital),Ji′nan 250022,China)

机构地区：[1]山东大学附属儿童医院(济南市儿童医院)呼吸介入科,济南250022

出　　处：《中华实用儿科临床杂志》2022年第24期1907-1910,共4页Chinese Journal of Applied Clinical Pediatrics

基　　金：山东省重特大疾病"防诊控治康"科技示范工程(2021SFGC0503)。

摘　　要：目的探讨儿童先天性气管狭窄(CTS)合并交通性支气管肺前肠畸形(CBPFM)的临床分型、诊断方法及治疗方案。方法对2021年5月、2022年1月于山东大学附属儿童医院呼吸介入科就诊的2例CTS合并CBPFM患儿的临床资料进行回顾性分析,并检索国内外数据库,总结该病的临床特征,探讨CBPFM的分型方法。结果例1,女,4岁,因"反复咳喘4年余"就诊,诊断:CBPFM(Ⅱ型,右侧);CTS。行外科胸腔镜右肺切除+食管修补术。例2,女,7个月2 d,因"进食、呼吸困难伴口唇发绀7个月"就诊,诊断:CBPFM(Ⅱ型,左侧);CTS。先后行滑动气管成形术,左肺切除术+食管修补术。共检索英文文献8篇,中文文献1篇。共收录CBPFM患儿21例,其中CTS合并CBPFM患儿12例,诊断证据充分的CTS均与ⅠA型、Ⅱ型CBPFM伴发。结论CTS合并CBPFM可导致患儿发生严重的喘息、呼吸困难,临床医师需加强认识,提高警惕。CTS与ⅠA型与Ⅱ型CBPFM两种畸形可能存在潜在联系,值得更深入研究。Objective To explore the clinical typing,diagnostic method and treatment plan of congenital tracheal stenosis(CTS)combined with communicating bronchopulmonary foregut malformation(CBPFM)in children.Methods The clinical data of 2 children with CTS and CBPFM who were treated in the Center for Respiratory Intervention of Children′s Hospital Affiliated to Shandong University in May 2021 and January 2022 were retrospectively analyzed.Studies were retrieved from domestic and foreign databases,so as to summarize the clinical characteristics of CTS complicated with CBPFM and investigate the typing method of CBPFM.Results One patient was a 4-year-old girl,who sought the medical advice due to"recurrent cough and asthma for more than 4 years".She was diagnosed with typeⅡCBPFM at the right side and CTS.Surgical thoracoscopic right pneumonectomy plus oesophageal repair was performed.The other patient was a 7-month-and-2-day-old female,who visited the hospital for"difficult eating,dyspnea and purple lip cyanosis for 7 months".This patient was diagnosed with typeⅡCBPFM at the left side and CTS.Slide tracheoplasty and left pneumonectomy+oesophageal repair were performed successively.Eight English and one Chinese studies were collected.Twenty-one children with CBPFM and 12 children with CTS and CBPFM were included.Eleven CTS cases with sufficient diagnostic evidence were complicated with typeⅠA and typeⅡCBPFM.Conclusions CTS and CBPFM can lead to severe wheezing and dyspnea.Clinicians should enhance their awareness and be more cautious.There may be a potential link between CTS and typeⅠA and typeⅡCBPFM,and further investigation is required.

关 键 词：气管狭窄 先天性 先天性交通性支气管肺前肠畸形 儿童 

分 类 号：R725.6[医药卫生—儿科]