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气管发育不全:对这一严重诊断的处理方法。病例报告和文献复习。

Tracheal agenesis: approach towards this severe diagnosis. Case report and review of the literature.

机构信息

Department of Pediatrics, Subdivision of Neonatology, VU University Medical Center, De Boelelaan 1117, 1081 HV Amsterdam, The Netherlands.

出版信息

Eur J Pediatr. 2012 Mar;171(3):425-31. doi: 10.1007/s00431-011-1563-x. Epub 2011 Sep 15.

DOI:10.1007/s00431-011-1563-x
PMID:21918809
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3284653/
Abstract

Tracheal agenesis (TA) is a severe congenital disorder with often an unexpected emergency presentation. There is complete or partial absence of the trachea below the larynx, with presence or absence of a tracheoesophageal fistula (TOF). A neonate with TA is described, and another 48 cases found in literature are reviewed. Due to absence of a TOF, five cases were diagnosed prenatally because of congenital high airway obstruction syndrome (CHAOS). When a TOF is present, polyhydramnion and several other congenital malformations seen on the ultrasound examination should alert clinicians of potential tracheal problems. Prenatal magnetic resonance imaging (MRI) may provide a definitive diagnosis. Postnatal diagnosis is based on recognition of specific clinical signs in the newborn with TA: respiratory distress with breathing movement without appropriate air entry, no audible cry, and failed endotracheal intubation. Despite progress in surgical interventions, mortality remains high. Prenatal diagnosis of TA is possible, but only if a TOF is absent resulting in CHAOS. Prenatal diagnosis of polyhydramnion and other congenital malformation should alert clinicians of potential tracheal problems. Prenatal MRI may provide a definitive diagnosis.

摘要

气管发育不全(TA)是一种严重的先天性疾病,常以意外急症的形式出现。TA 是指喉部以下的气管完全或部分缺失,同时可能伴有或不伴有气管食管瘘(TOF)。本文描述了一例 TA 新生儿病例,并对文献中发现的另外 48 例病例进行了回顾。由于不存在 TOF,5 例病例因先天性高位气道梗阻综合征(CHAOS)而在产前被诊断。当存在 TOF 时,超声检查中应注意羊水过多和其他几种先天性畸形,这些畸形可能提示存在潜在的气管问题。产前磁共振成像(MRI)可能提供明确的诊断。产后诊断基于对具有 TA 的新生儿特定临床体征的识别:呼吸窘迫伴呼吸运动但无适当的空气进入,无可闻哭声,且气管插管失败。尽管外科干预取得了进展,但死亡率仍然很高。如果不存在导致 CHAOS 的 TOF,则可以进行产前诊断 TA,但这仅在少数情况下可行。产前诊断羊水过多和其他先天性畸形应提示临床医生注意潜在的气管问题。产前 MRI 可能提供明确的诊断。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/82aa6e41a736/431_2011_1563_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/819cbfe38a16/431_2011_1563_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/57b1a525c4f6/431_2011_1563_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/82aa6e41a736/431_2011_1563_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/819cbfe38a16/431_2011_1563_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/57b1a525c4f6/431_2011_1563_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af1d/3284653/82aa6e41a736/431_2011_1563_Fig3_HTML.jpg

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