生物胶封堵治疗儿童先天性气管胆管瘘1例并文献复习

Congenital tracheobiliary fistula treated with biological glue plugging under bronchoscopic guidance： a case report and literature review

目的探讨儿童先天性气管胆管瘘（CTBF）的临床特点及诊疗方法。方法选取湖北省妇幼保健院儿童呼吸内科2016年收治的儿童CTBF 1例，并进行相关文献复习，分析该病的临床特点、诊断方法、治疗现状及临床转归。结果本例患儿年龄3岁7个月，以反复咳嗽、肺炎、肺不张为主要临床表现，支气管镜下碘油造影诊断为CTBF，并经支气管镜下生物胶封堵成功治愈。迄今英文文献中仅有30例儿童CTBF被报道，2例中文文献报道。主要表现为咳嗽、呼吸困难、胆汁痰或呕吐、吸入性肺炎、肺不张或肺气肿等，诊断方法包括支气管镜、支气管造影、胆道造影、肝胆核素扫描、CT扫描和磁共振成像等。除本例患儿采用支气管镜下生物胶封堵瘘管外，其余患儿均采用手术治疗，手术方式包括瘘管结扎术、胃造口术、肝切除术、瘘管-空肠Roux-en-Y吻合术、肝门-空肠吻合术、胆囊-空肠吻合术等。4例死亡，余均痊愈。结论儿童持续性慢性咳嗽经常规治疗控制不佳时，应考虑CTBF的可能，应及时行支气管镜检查以明确诊断。支气管镜下碘油造影是一种简单可行的诊断方法，除手术治疗外，支气管镜下肺泡灌洗和生物胶封堵对治疗未并严重胆道畸形的儿童CTBF是一种有效的方式。

ObjectiveTo investigate the clinical characteristics, diagnosis and treatment of congenital tracheobiliary fistula （CTBF） in children.MethodsA case of CTBF admitted into the Department of Pediatric Respiration, Hubei Maternal and Child Health Care Hospital in 2016 was reported, and the related literatures were reviewed.The clinical features, diagnostic methods, treatment status and clinical outcomes of the disease were analyzed.ResultsThe patient was 3 years and 7 months old.The main clinical manifestations were recurrent cough, pneumonia and atelectasis.CTBF was diagnosed by means of iodine oil radiography and treated with biological glue plugging under bronchoscopic guidance.So far, only 30 cases of CTBF have been reported in the English literatures, but only 2 cases in the Chinese literatures.The main clinical manifestations were cough, dyspnea, sputum or bile vomiting, aspiration pneumonia, atelectasis or emphysema.Diagnostic methods for CTBF included bronchoscopy, bronchial angiography and cholangiography, hepatobiliary scan, CT scan and magnetic resonance imaging.Except for this case treated with biological glue plugging under bronchoscopic guidance, all other patients were treated with surgery.The operation methods included fistula ligation, gastrostomy, liver resection, fistula jejunum Roux-en-Y anastomosis, hepatic hilum jejunum anastomosis, gallbladder jejunum anastomosis, etc.Only 4 cases died, and the rest of the patients recovered.ConclusionsCTBF should be suspected in children with persistent chronic cough.Iodine oil radiography through bronchoscopy is a simple and feasible method for diagnosis of CTBF.Besides surgery, the lavage and the biological glue plugging method through bronchoscopy is also an effective way to treat CTBF in children without severe biliary malformation.