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双重 H 型气管食管瘘;为何不能同期修复?病例报告及文献复习。

A dual H-type tracheoesophageal fistula; why not being repaired simultaneously? A case report and review of literature.

机构信息

Pediatric Gastroenterology, Hepatology and Nutrition Research Center, Research Institute for Children's Health, Shahid Beheshti University of Medical Sciences, Tehran, Iran.

Department of Pediatrics, School of medicine, Kerman University of Medical Sciences, Kerman, Iran.

出版信息

BMC Pediatr. 2023 Jun 19;23(1):308. doi: 10.1186/s12887-023-03945-y.

DOI:10.1186/s12887-023-03945-y
PMID:37337161
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10278301/
Abstract

BACKGROUND

H-type Tracheoesophageal Fistula (TEF) is a particular type of congenital esophageal anomalies, in which patients present with non-specific symptoms that can result in delayed diagnosis. Here, we report two pediatric cases with a rarer variant called ‟dual H-type TEFˮ.

CASE PRESENTATION

We present two cases of H-type TEF. The first was a 45-day-old boy with feeding problem and cyanosis while feeding, and the second was a three-month-old girl with cough and choking after feeding from the first day of birth. In both cases, two separate TEFs were detected during diagnostic evaluation by flexible bronchoscopy. Both were repaired simultaneously through a cervical incision. The first patient deteriorated 13 days after the surgery, disturbancing in acid-base balance and expired unfortunately.

CONCLUSION

Hence, it is necessary to consider the possibility of double TEF in any newly diagnosed H-type TEF.

摘要

背景

H 型气管食管瘘(TEF)是一种特殊类型的先天性食管畸形,患者表现出非特异性症状,可能导致诊断延迟。在此,我们报告两例罕见的“双 H 型 TEF”儿科病例。

病例介绍

我们报告两例 H 型 TEF。第一例是一名 45 天大的男孩,在喂养时出现喂养问题和发绀,第二例是一名三个月大的女孩,从出生第一天开始喂养后就出现咳嗽和呛咳。在这两种情况下,通过软性支气管镜检查在诊断评估中均检测到两个独立的 TEF。均通过颈部切口同时修复。第一例患者在手术后 13 天恶化,酸碱平衡紊乱,不幸去世。

结论

因此,对于任何新诊断的 H 型 TEF,都有必要考虑双重 TEF 的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/51ac474f3af5/12887_2023_3945_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/2acff5abb0a7/12887_2023_3945_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/3a858a33fca6/12887_2023_3945_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/51ac474f3af5/12887_2023_3945_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/2acff5abb0a7/12887_2023_3945_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/3a858a33fca6/12887_2023_3945_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e5a/10278301/51ac474f3af5/12887_2023_3945_Fig3_HTML.jpg

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Laryngotracheoesophageal cleft, a rare differential diagnosis of esophageal atresia.喉气管食管裂,一种罕见的食管闭锁鉴别诊断。
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Congenital H-type tracheoesophageal fistula: A multicenter review of outcomes in a rare disease.先天性H型气管食管瘘:一种罕见疾病结局的多中心综述
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Long-term outcomes following H-type tracheoesophageal fistula repair in infants.婴儿H型气管食管瘘修复术后的长期预后
Pediatr Surg Int. 2017 Feb;33(2):187-190. doi: 10.1007/s00383-016-4012-0. Epub 2016 Nov 28.
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Laryngotracheoesophageal clefts.喉气管食管裂
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