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唇腭裂合并气管支气管软化症患者:一例报告及日本文献综述

Cleft-lip-plate patient with tracheobronchomalacia: A case report and review of the literature in Japan.

作者信息

Hitosugi Takashi, Mitsuyasu Takeshi, Yokoyama Takeshi

机构信息

Department of Dental Anesthesiology, Faculty of Dental Science, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan.

Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic and Surgical Sciences. Faculty of Dental Science, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan.

出版信息

JPRAS Open. 2020 Sep 30;26:60-68. doi: 10.1016/j.jpra.2020.09.003. eCollection 2020 Dec.

DOI:10.1016/j.jpra.2020.09.003
PMID:33163607
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7609487/
Abstract

BACKGROUND

Tracheobronchomalacia (TBM) is a severe life-threatening perioperative complication. It is a rare condition caused by congenital and developmental anomalies of the trachea and/or the bronchus. It is often difficult to diagnose TBM before surgery as this congenital disease presents very few symptoms preoperatively and most often appears postoperatively.

CASE PRESENTATION AND REVIEW

The study describes a case of cleft-lip-plate (CL/P) in a 7-month-old Japanese female with TBM and Tetralogy of Fallot syndrome. Before undergoing cleft-lip surgery, her TBM was not fully elucidated by preoperative examinations, and the operation was completed uneventfully. After the surgery, however, she started showing severe respiratory distress and developed hypoxia and bradycardia in the operating room. CPR was performed successfully, but a bronchoscopy revealed a severely collapsed airway, and the pathological condition was diagnosed as TBM occurred postoperatively. Eight months later, she died of sudden respiratory failure similar to that of the postoperative event caused by TBM. A literature review was conducted on the complications of CL/P from 1990 to 2017 in Japan.

CONCLUSIONS

It was hypothesized that CL/P with congenital heart disease (CHD) and TBM with CHD may crossover in relatively high rates. Currently, there are very few solutions available to treat severe airway obstruction related to TBM. This highlights the need for preoperative diagnosis of TBM as an important step in overcoming severe airway complications.

摘要

背景

气管支气管软化症(TBM)是一种严重危及生命的围手术期并发症。它是由气管和/或支气管的先天性和发育异常引起的罕见病症。术前通常很难诊断TBM,因为这种先天性疾病术前症状很少,且大多在术后出现。

病例报告与回顾

该研究描述了一名7个月大患有TBM和法洛四联症的日本女性唇腭裂(CL/P)病例。在进行唇裂手术前,术前检查未能完全明确其TBM情况,手术顺利完成。然而,术后她开始出现严重呼吸窘迫,并在手术室出现缺氧和心动过缓。心肺复苏成功实施,但支气管镜检查显示气道严重塌陷,病理状况被诊断为术后发生的TBM。八个月后,她死于类似于由TBM引起的术后事件的突发呼吸衰竭。对1990年至2017年日本CL/P的并发症进行了文献综述。

结论

据推测,患有先天性心脏病(CHD)的CL/P和患有CHD的TBM可能有较高的交叉发生率。目前,治疗与TBM相关的严重气道阻塞的方法非常有限。这凸显了术前诊断TBM作为克服严重气道并发症重要步骤的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/dbace7e1993a/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/b1ee1ffc88f5/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/3b2ca2067fc8/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/c4db930ac4ab/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/dbace7e1993a/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/b1ee1ffc88f5/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/3b2ca2067fc8/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/c4db930ac4ab/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9a49/7609487/dbace7e1993a/gr4.jpg

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本文引用的文献

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The Prevalence of Congenital Heart Disease in Nonsyndromic Cleft Lip and/or Palate: A Systematic Review of the Literature.非综合征性唇裂和/或腭裂患者先天性心脏病的患病率:文献系统评价
Ann Plast Surg. 2017 Aug;79(2):214-220. doi: 10.1097/SAP.0000000000001069.
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Outcomes of Extracorporeal Life Support During Surgery for the Critical Airway Stenosis.重症气道狭窄手术期间体外生命支持的结果
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[Perioperative Management of a Child with Vocal Adhesion Leading to Unexpected Difficult Airway].
Masui. 2016 Jun;65(6):590-3.
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Paediatric Tracheomalacia.小儿气管软化症
Paediatr Respir Rev. 2016 Jan;17:9-15. doi: 10.1016/j.prrv.2015.03.002. Epub 2015 Mar 17.
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Updating the epidemiology of cleft lip with or without cleft palate.更新唇裂伴或不伴腭裂的流行病学。
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Prevalence at birth of cleft lip with or without cleft palate: data from the International Perinatal Database of Typical Oral Clefts (IPDTOC).唇裂伴或不伴腭裂的出生患病率:来自国际典型口腔裂隙围产期数据库(IPDTOC)的数据。
Cleft Palate Craniofac J. 2011 Jan;48(1):66-81. doi: 10.1597/09-217. Epub 2010 Apr 6.
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Influence of tracheobronchomalacia on outcome of surgery in children with congenital heart disease and its management.气管支气管软化症对先天性心脏病患儿手术结果的影响及其处理。
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Tracheomalacia and bronchomalacia in children: incidence and patient characteristics.儿童气管软化症和支气管软化症:发病率及患者特征
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Tracheomalacia and tracheobronchomalacia in children and adults: an in-depth review.儿童和成人的气管软化症及气管支气管软化症:深入综述
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The stove-in chest with paradoxical respiration.伴有反常呼吸的胸部凹陷。
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