切换至 "中华医学电子期刊资源库"
高级检索
中华妇幼临床医学杂志(电子版)

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
全文 ( ) 下载PDF ( ) 导出引用
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]. 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]. 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
[1]
Fallon SC, Langer JC, St Peter SD, et al. Congenital H-type tracheoesophageal fistula: a multicenter review of outcomes in a rare disease [J]. J Pediatr Surg, 2017, 52(11): 1711-1714. DOI: 10.1016/j.jpedsurg.2017.05.002.
[2]
钟微，李乐，郑珊，等. 先天性食管闭锁诊断及治疗(专家共识) [J]. 中华小儿外科杂志，2014, 35(8): 623-626. DOI: 10.3760/cma.j.issn.0253-3006.2014.08.016.
[3]
Tiwari C, Nagdeve N, Saoji R, et al. Congenital H-type tracheo-oesophageal fistula: An institutional review of a 10-year period [J]. J Mother Child, 2021, 24(4): 2-8. DOI: 10.34763/jmotherandchild.20202404.d-20-00004.
[4]
Stavroulias D, Ampollini L, Carbognani P, et al. Late presentation of congenital H-type tracheoesophageal fistula in an immunocompromised patient [J]. Eur J Cardiothorac Surg, 2011, 40(2): e98-e100. DOI: 10.1016/j.ejcts.2011.03.026.
[5]
李云荣，潘征夏. 小儿先天性H型气管食管瘘的诊断与治疗[J].临床小儿外科杂志，2016, 15(6): 583-586. DOI: 10.3969/j.issn.1671-6353.2016.06.016.
[6]
田俊. 气管食管瘘的诊断治疗进展 [J]. 中华耳鼻咽喉头颈外科杂志，2022, 57(4): 524-530. DOI: 10.3760/cma.j.cn115330-20210925-00630,
[7]
Leven NL, Varco RL, Lannin BG, et al. The surgical management of congenital atresia of the esophagus and tracheo-esophageal fistula [J]. Ann Surg, 1952, 136(4): 701-719.
[8]
Babbitt DP. Double tracheoesophageal fistula without atresia; report of a case [J]. N Engl J Med, 1957, 257(15): 713-714. DOI: 10.1056/NEJM195710102571506
[9]
Benjamin B, Pham T. Diagnosis of H-type tracheoesophageal fistula [J]. J Pediatr Surg, 1991, 26(6): 667-671. DOI: 10.1016/0022-3468(91)90007-g.
[10]
Schulte T, Ankermann T, Claas A, et al. An extremely rare abnormality of a double tracheoesophageal fistula without atresia of the esophagus; a case report and review of the literature [J]. J Pediatr Surg, 2009, 44(10): e9-e12. DOI: 10.1016/j.jpedsurg.2009.06.032.
[11]
Mattei P. Double H-type tracheoesophageal fistulas identified and repaired in 1 operation [J]. J Pediatr Surg, 2012, 47(11): e11-e13. DOI: 10.1016/j.jpedsurg.2012.06.012.
[12]
Oosthuizen JC, Ramli RR, Aworanti OM, et al. Trans-cervical, trans-tracheal approach to proximal tracheo-oesophageal fistula: a novel technique [J]. Int J Pediatr Otorhinolaryngol, 2014, 78(9): 1554-1556. DOI: 10.1016/j.ijporl.2014.06.017.
[13]
De Carolis MP, Cocca C, Cota F, et al. Double tracheoesophageal fistula and azygos lobe [J]. Pediatr Neonatol, 2016, 57(6): 546-547. DOI: 10.1016/j.pedneo.2016.02.006.
[14]
Hosseini A, Sinaei R, Yeganeh MH, et al. A dual H-type tracheoesophageal fistula; why not being repaired simultaneously? A case report and review of literature [J]. BMC Pediatr, 2023, 23(1): 308. DOI: 10.1186/s12887-023-03945-y.
[15]
Wong MD, Thomas RJ, Powell J, et al. Flexible bronchoscopy diagnosis of uncommon congenital h-type tracheoesophageal fistula, dual fistulae, bronchoesophageal fistula, and recurrence of fistula in children: a 20-year experience [J]. J Bronchol Interv Pulmonol, 2022, 29(2): 99-108. DOI: 10.1097/LBR.0000000000000793.
[16]
赵家伟，赵勇，华凯云，等. 先天性双气管食管瘘的诊治一例并文献复习[J]. 中华小儿外科杂志，2022, 43(2): 122-126. DOI: 10.3760/cma.j.cn421158-20200909-00602.
[17]
Gutierrez RS, Guelfand M, Balbontin PV. Congenital and acquired tracheoesophageal fistulas in children [J]. Semin Pediatr Surg, 2021, 30(3): 151060. DOI: 10.1016/j.sempedsurg.2021.151060.
[18]
Cuestas G, Rodríguez V, Millán C, et al. H-type tracheoesophageal fistula in the neonatal period: difficulties in diagnosis and different treatment approaches. A case series [J]. Arch Argent Pediatr, 2020, 118(1): 56-60. DOI: 10.5546/aap.2020.eng.56.
[19]
Al-Salem AH, Mohaidly MA, Al-Buainain HM, et al. Congenital H-type tracheoesophageal fistula: a national multicenter study [J]. Pediatr Surg Int, 2016, 32(5): 487-491. DOI: 10.1007/s00383-016-3873-6.
[20]
Shaw-Smith C. Oesophageal atresia, tracheo-oesophageal fistula, and the VACTERL association: review of genetics and epidemiology [J]. J Med Genet, 2006, 43(7): 545-554. DOI: 10.1136/jmg.2005.038158.
[21]
Spitz L. Oesophageal atresia [J]. Orphanet J Rare Dis, 2007, 2: 24. DOI: 10.1186/1750-1172-2-24.
[22]
Perry M, Eick J, Jakob K, et al. Clinical images - a quarterly column: early presentation of H-type tracheoesophageal fistula[J]. Ochsner J, 2013，13(4)：483-485.
[23]
Goyal A, Potter F, Losty PD. Transillumination of H-type tracheoesophageal fistula using flexible miniature bronchoscopy: an innovative technique for operative localization [J]. J Pediatr Surg, 2005, 40(6): e33-e34. DOI: 10.1016/j.jpedsurg.2005.03.029.
[24]
王忠弢，石永生，王永军，等. 现代儿科呼吸介入技术在气管食管瘘中的应用进展 [J]. 中国实用儿科杂志，2020, 35(10): 779-783. DOI: 10.19538/j.ek2020100609.
[25]
马永钰，杨仕武，王舒钰，等. 不同术式治疗婴儿食管裂孔疝的疗效分析[J/OL]. 中华妇幼临床医学杂志(电子版), 2024, 20(1): 25-31. DOI：10.3877/cma.j.issn.1673-5250.2024.01.004.
[26]
Taghavi K, Tan Tanny SP, Hawley A, et al. H-type congenital tracheoesophageal fistula: Insights from 70 years of the Royal Children′s Hospital experience [J]. J Pediatr Surg, 2021, 56(4): 686-691. DOI: 10.1016/j.jpedsurg.2020.06.048.
[27]
Parolini F, Morandi A, Macchini F, et al. Cervical/thoracotomic/thoracoscopic approaches for H-type congenital tracheo-esophageal fistula: a systematic review [J]. Int J Pediatr Otorhinolaryngol, 2014, 78(7): 985-989. DOI: 10.1016/j.ijporl.2014.04.011.
[28]
Luscan R, Simon F, Khen Dunlop N, et al. Thulium LASER for endoscopic closure of tracheoesophageal fistula in esophageal atresia′s spectrum: an appropriate tool? [J]. J Pediatr Surg, 2021, 56(10): 1752-1756. DOI: 10.1016/j.jpedsurg.2020.10.005.
[29]
Lelonge Y, Varlet F, Varela P, et al. Chemocauterization with trichloroacetic acid in congenital and recurrent tracheoesophageal fistula: a minimally invasive treatment [J]. Surg Endosc, 2016, 30(4): 1662-1666. DOI: 10.1007/s00464-015-4352-1.
[30]
徐慧姣，陈佳俊，赖冠宇，等. 先天性食管裂孔疝患儿临床诊疗研究 [J/OL]. 中华妇幼临床医学杂志(电子版), 2024, 20(1): 32-38. DOI: 10.3877/cma.j.issn.1673-5250.2024.01.005.
[31]
魏延栋，王莹，马立霜，等. 胸腔镜手术治疗低出生体质量儿Ⅲ型食管闭锁经验分析[J]. 发育医学电子杂志，2023, 11(4): 277-281. DOI: 10.3969/j.issn.2095-5340.2023.04.006.
[32]
Gregory S, Chun RH, Parakininkas D, et al. Endoscopic esophageal and tracheal cauterization for closure of recurrent tracheoesophageal fistula: a case report and review of the literature [J]. Int J Pediatr Otorhinolaryngol, 2017, 98: 158-161. DOI: 10.1016/j.ijporl.2017.04.051.
[33]
Nardo GD, Oliva S, Barbato M, et al. Video: argon plasma coagulator in a 2-month-old child with tracheoesophageal fistula [J]. Surg Endosc, 2012, 26(9): 2678-2680. DOI: 10.1007/s00464-012-2214-7.
[1] 李浩纶, 杨佳麒, 李羽. 盐酸氢吗啡酮在小儿术后镇痛作用中的研究现状[J]. 中华妇幼临床医学杂志(电子版), 2024, 20(02): 166-171.
[2] 刘清, 汪志凌. 肠道真菌与儿童炎症性肠病[J]. 中华妇幼临床医学杂志(电子版), 2024, 20(02): 172-178.
[3] 王佳璇, 黄立渠, 郭云飞. 膀胱颈悬吊术在儿童神经源性尿失禁治疗的研究现状[J]. 中华妇幼临床医学杂志(电子版), 2024, 20(02): 179-184.
[4] 吴茗, 朱芬华, 刘丹如, 俞晔珩, 林彤, 李健. 儿童GNPTAB/GNPTG基因突变致黏脂贮积症2例并文献复习[J]. 中华妇幼临床医学杂志(电子版), 2024, 20(02): 185-191.
[5] 郑伟军, 方一凡, 吴典明, 王翔, 陈飞, 刘明坤. 先天性肠旋转不良诊治分析：单中心10年经验总结[J]. 中华普外科手术学杂志(电子版), 2024, 18(03): 338-341.
[6] 李盼, 韦登飞, 乔克坤, 李熊刚. 超声引导下骶管阻滞与髂腹股沟及髂腹下神经阻滞用于小儿腹腔镜疝囊高位结扎术的比较[J]. 中华疝和腹壁外科杂志(电子版), 2024, 18(03): 326-330.
[7] 成紫琳, 戴明, 李建华, 马靓. 加速康复外科理念在儿童腹股沟疝围手术期的应用[J]. 中华疝和腹壁外科杂志(电子版), 2024, 18(03): 331-335.
[8] 钟知足, 陶波圆, 曾纪晓, 吴强, 王哲, 余家康, 刘斐, 徐晓钢, 兰梦龙, 梁子建, 李燕秋, 刘凤君. 儿童腹腔镜胆总管囊肿根治术后并发症危险因素分析[J]. 中华腔镜外科杂志(电子版), 2024, 17(02): 83-89.
[9] 曾纪晓, 梁子建. 单孔腹腔镜手术在儿童普通外科中的应用与展望[J]. 中华腔镜外科杂志(电子版), 2024, 17(02): 65-69.
[10] 王增萌, 彭春辉, 吴东阳, 王凯, 闫俊, 黄心洁, 陈亚军. 先天性胆总管囊肿术后吻合口狭窄/肝内胆管结石的腹腔镜再手术经验[J]. 中华腔镜外科杂志(电子版), 2024, 17(02): 111-115.
[11] 张秩坤, 李中策, 郑奕菲, 戚士芹. 经脐单孔腹腔镜脾部分切除在儿童脾病中的应用体会[J]. 中华腔镜外科杂志(电子版), 2024, 17(02): 116-119.
[12] 刘宇立, 姚瑶, 徐锦雯. 过敏性紫癜性肾炎患儿肾功能不全的影响因素分析[J]. 中华肾病研究电子杂志, 2024, 13(02): 68-73.
[13] 汪久宏, 丁莉, 梁丽. 腹部彩超联合高频彩超用于小儿肠套叠诊断经验及价值[J]. 中华消化病与影像杂志(电子版), 2024, 14(03): 226-228.
[14] 轩欢欢, 刘凤麟, 李伟, 李自普, 贾宝俊, 王金菊, 满宜刚. 儿童川崎病休克综合征合并可逆性胼胝体压部病变综合征的诊断学特征并文献复习[J]. 中华诊断学电子杂志, 2024, 12(02): 95-100.
[15] 刘兆全, 张芳芳, 宋洪浩, 王刚, 崔明宇. 儿童腹腔炎性肌纤维母细胞瘤的诊断学特征并文献复习[J]. 中华诊断学电子杂志, 2024, 12(02): 101-106.
阅读次数
全文


摘要

版权所有 中华医学会 中华医学电子音像出版社有限责任公司  京ICP备14006079号-1  网络出版服务许可证：(署)网出证(京)字第075号