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中华妇幼临床医学杂志(电子版) ›› 2024, Vol. 20 ›› Issue (02) : 216 -223. doi: 10.3877/cma.j.issn.1673-5250.2024.02.013

论著

双H型先天性气管食管瘘2例并文献复习
刘芸1, 余宏川1, 孙欣荣1, 陈伟超1, 杨婷婷1,†()   
  1. 1. 西安市儿童医院呼吸一科，西安　710003
  • 收稿日期:2023-10-01 修回日期:2024-02-09 出版日期:2024-04-01
  • 通信作者: 杨婷婷

Double H-type congenital tracheoesophageal fistulas: two cases report and literature review

Yun Liu1, Hongchuan Yu1, Xinrong Sun1, Weichao Chen1, Tingting Yang1,†()   

  1. 1. Department of Respiratory, Xi′an Children′s Hospital, Xi′an 710003, Shaanxi Province, China
  • Received:2023-10-01 Revised:2024-02-09 Published:2024-04-01
  • Corresponding author: Tingting Yang
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引用本文：

刘芸, 余宏川, 孙欣荣, 陈伟超, 杨婷婷. 双H型先天性气管食管瘘2例并文献复习[J/OL]. 中华妇幼临床医学杂志(电子版), 2024, 20(02): 216-223.

Yun Liu, Hongchuan Yu, Xinrong Sun, Weichao Chen, Tingting Yang. Double H-type congenital tracheoesophageal fistulas: two cases report and literature review[J/OL]. Chinese Journal of Obstetrics & Gynecology and Pediatrics(Electronic Edition), 2024, 20(02): 216-223.

目的

探讨双H型先天性气管食管瘘(DCTEFs)患儿的临床特点。

方法

选择西安市儿童医院收治的2例DCTEFs患儿(患儿1、2)为研究对象。采用回顾性分析方法，对这2例患儿的临床资料、诊疗方案进行分析。以"气管食管瘘""H型气管食管瘘""双气管食管瘘""双H型气管食管瘘""V型食管闭锁""单纯性气管食管瘘""食管气管瘘"为中文主题词，在中国知网、万方数据库中进行文献检索；再以"tracheoesophageal fistula""H-type tracheoesophageal fistula""double H-type tracheoesophageal fistula""double tracheoesophageal fistula""Ⅴ-type esophageal atresia"为英文主题词，在PubMed、Embase等英文数据库中进行文献检索。检索时间设定为1952年1月至2022年11月。

结果

①患儿1(4个月2 d龄)以生后呛奶、吐奶、反复呼吸道感染为主要表现，于2018年8月29日入本院治疗。患儿2(2岁11个月)以生后呛咳伴呕吐、反复呼吸道感染为主要表现，于2020年3月24日入本院治疗。对2例患儿进行支气管镜检查均发现2个H型气管食管瘘，并进行内镜下气管食管瘘封堵术(TFC)，使用氩气刀灼烧食管内侧瘘口周围黏膜，患儿1治愈，患儿2于术后8个月复发。②根据本研究设定的检索策略，共检索出10篇相关文献，纳入共计10例DCTEFs患儿诊疗研究。其中，7例患儿(患儿4、6～10、12)临床病例资料相对完整，临床表现均为进食后发绀、呛咳、吐奶或腹胀等呼吸道及消化道症状，1例(患儿12)有反复呼吸道感染史。这10例患儿中，仅1例(患儿5)的食管造影术结果显示2个瘘口，对3例(患儿7、9、12)采取支气管镜检查均发现2个瘘口，对1例(患儿11)采取支气管镜联合胃镜检查发现2个瘘口，1例(患儿3)尸检发现2个瘘口，1例(患儿4)尸检发现第2个瘘口，3例(患儿6、8、10)在术中或术后均发现第2个瘘口。4例(患儿6～8、10)采取开放性手术治愈，1例(患儿12)采取胸腔镜手术治愈。

结论

DCTEFs是一种罕见病，该病患儿临床表现无特异性，易被临床漏诊，检查过程中应仔细寻找第2瘘管，一旦确诊，应及时采取手术治疗措施。

关键词: 气管食管瘘，先天性, 双H型气管食管瘘, 支气管镜, 气管食管瘘封堵术, 儿童
Objective

To investigate the clinical characteristics of children with double H-type congenital tracheoesophageal fistulas (DCTEFs).

Methods

Two cases of DCTEFs (patient 1 and 2)treated at Xi′an Children′s Hospital were selected in this study. A retrospective analysis was conducted to examine the clinical data, diagnostic methods, and treatment plans for these two patients. Literature searches were performed in Chinese databases such as CNKI and Wanfang using Chinese keywords including " tracheoesophageal fistula" " H-type tracheoesophageal fistula" " double tracheoesophageal fistula" " double H-type tracheoesophageal fistula" " V-type esophageal atresia" " simple tracheoesophageal fistula" and " esophageal tracheal fistula". Additionally, English databases in PubMed and Embase were searched using English keywords such as " tracheoesophageal fistula" " H-type tracheoesophageal fistula" " double H-type tracheoesophageal fistula" " double tracheoesophageal fistula" and " V-type esophageal atresia". The search period was set from January 1952 to November 2022.

Results

① Patient 1 (4 months and 2 days old) was admitted to our hospital on August 29, 2018, with symptoms of choking on milk, vomiting, and recurrent respiratory infections after birth. Patient 2 (2 years and 11 months old) was admitted on March 24, 2020, with symptoms of choking cough, vomiting, and recurrent respiratory infections after birth. Both patients underwent bronchoscopy and were found to have two H-type tracheoesophageal fistulas. They both underwent endoscopic tracheoesophageal fistula closure (TFC), with argon beam coagulation of the mucosa around the inner fistula of the esophagus. Patient 1 was cured, while patient 2 experienced a recurrence 8 months after surgery. ② According to the search strategies established in this study, a total of 10 relevant articles were retrieved, involving 10 cases with DCTEFs. Among them, 7 patients (patients 4, 6-10, 12) had relatively complete clinical case data, all showing respiratory and gastrointestinal symptoms, such as cyanosis after feeding, choking cough, vomiting, or abdominal distension. One patient (patient 12) had a history of recurrent respiratory infections. Among the 10 patients, only one patient (patient 5) had two fistulas revealed by esophagography, three patients (patient 7, 9, 12) were found to have two fistulas by bronchoscopy, one patient (patient 11) was found to have two fistulas by combined bronchoscopy and gastroscopy, one patient (patient 3) had two fistulas discovered at autopsy, one patient (patient 4) had the second fistula discovered at autopsy, and three patients (patients 6, 8, 10) had the second fistula discovered during or after surgery. Four patients (patients 6-8, 10) were cured by open surgery, and one patient (patient 12) was cured by thoracoscopic surgery.

Conclusions

DCTEFs is a rare disease with nonspecific clinical manifestations, prone to missed diagnosis. Careful search for the second fistula during examination is essential, and once diagnosed, surgical treatment should be performed promptly.

Key words: Tracheoesophageal fistula, congenital, Double H-type tracheoesophageal fistula, Bronchoscopy, Tracheoesophageal fistula closure, Child
图1　DCTEFs患儿1(男性，4个月2 d龄)支气管镜检查结果[图1A、1B：声门下气管上段膜部偏右侧可见2个瘘口(C8～T1)，右主支气管狭窄，镜体不能通过，左主支气管稍狭窄(黑色箭头所示)]　 图2　DCTEFs患儿2(女性，2岁11个月龄)支气管镜检查结果[气管上段可见2个瘘口(C8～T1)，瘘口内可见较多分泌物涌出(黑色箭头所示)]注：DCTEFs为双H型先天性气管食管瘘
表1 文献检索涉及的10例DCTEFs患儿及2例本研究纳入的DCTEFs患儿的临床特征比较
患儿编号 文献(第1作者，发表年) 性别 诊断年龄 临床表现 诊断 治疗方式及定位 预后及转归
影像学 食管造影 支气管镜
1 本研究 男 4个月2 d 呛奶、吐奶 胸部常规CT提示双肺内散在斑片影及实变 － 2个瘘口位于声门下气管上段膜部偏右侧 内镜下TFC 治愈
2 本研究 女 2岁11个月 呛咳、呕吐 胸部＋喉部常规CT及三维重建提示右肺上叶少许炎症 － 2个瘘口位于气管上段 内镜下TFC(无定位) 术后8个月余瘘复发
3 Leven等[7]，　1952 女 死亡后 － － － － － 死亡
4 Babbitt等[8]，1957 男 死亡后 进食后发绀、呕吐、腹胀 胸部X射线摄片示右肺中上叶无菌性肺炎 颈部可见1个瘘口 1个直径为3 mm瘘口位于气管下1/3处 开胸TFC(无定位) 死亡
5 Benjamin等[9]，　1991 － － － － 2个瘘口 － － －
6 Schulte等[10]，　2009 男 生后20 d 呕吐、呛咳 胸部X射线摄片示右上肺叶炎症浸润 T2下方1个瘘口 1个瘘口位于距隆突上方1.5 cm处 开胸TFC远端瘘管＋瘘管切断缝合(支气管镜) 治愈
7 Mattei等[11]，　2012 男 新生儿期 呛咳 胸部X射线摄片未见明显异常 胸内TEF 远、近端瘘口分别位于距隆突上方2 cm与气管上段 开胸TFC＋经右颈部TFC＋瘘管切断缝合(Fogarty气囊导管) 治愈
8 Oosthuizen等[12]，　2014 女 生后18 d 呼吸困难 胸部X射线摄片提示左下肺不张 阴性 1个瘘口位于气管下段 开胸TFC远端瘘管＋单纯结扎(支气管镜) 治愈
9 Carolis等[13]，2016 － － 呼吸困难、发绀、咳嗽 胸部常规CT示右肺散在斑片影、实变 － 2个瘘口位于颈段气管内 － －
10 Sharman等[14]，2020 女 生后14 d 体重不增、发绀 － 1个瘘口 阴性 经右颈部入TFC＋瘘管切断缝合＋填充组织(支气管镜) 治愈
11 Matthew等[15]，2021 男 13岁 － － － 联合胃镜发现2个瘘口 开胸TFC(无定位) －
12 赵家伟等[16]，　2022 女 4个月17 d 呛咳、腹胀 胸部CT及气管重建示T3水平气管后壁与食管前壁交通 T3水平可见1个瘘口 远、近端瘘口分别位于隆突上方1 cm与1.5 cm 胸腔镜TFC＋瘘管切断缝合(支气管镜导丝) 治愈
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