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伴有罕见相关血管和肛肠畸形的食管肺。

Esophageal lung with rare associated vascular and anorectal malformations.

作者信息

Alsaadi Ali, Alsufiani Hamdi A, Allugmani Mohammad D, Gora Altaf Hussain

机构信息

Department of Radiology, Madina Maternity and Children's Hospital, Madinah, Saudi Arabia.

Department of Pediatric Pulmonary, Madina Maternity and Children's Hospital, Madinah, Saudi Arabia.

出版信息

Radiol Case Rep. 2018 Feb 28;13(2):444-448. doi: 10.1016/j.radcr.2018.01.029. eCollection 2018 Apr.

DOI:10.1016/j.radcr.2018.01.029
PMID:29904493
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6000028/
Abstract

Esophageal lung is a rare communicating bronchopulmonary foregut malformation in which the main bronchus arising from the trachea is absent. The affected lung is usually hypoplastic and aerated via an anomalous airway originating from the esophagus. Other anomalies such as esophageal atresia with tracheoesophageal fistula or VACTERL (vertebral defects, anal atresia, cardiac defects, tracheoesophageal fistula, renal anomalies, and limb abnormalities) association can co-exist. The initial radiographic findings may be normal, but subsequent imaging usually shows progressive and recurrent lung collapse, probably because of recurrent aspiration through the anomalous airway and poor compliance of the affected lung during breathing. In this report, we describe a neonate with esophageal lung and rare associated anomalies, including anorectal malformation, pulmonary artery sling, and inferior vena cava interruption with azygous continuation. To our knowledge, this is the first report of esophageal lung with such associations.

摘要

食管肺是一种罕见的交通性支气管肺前肠畸形,其中起源于气管的主支气管缺如。患侧肺通常发育不全,并通过起源于食管的异常气道进行通气。其他异常情况,如食管闭锁合并气管食管瘘或VACTERL(脊柱缺陷、肛门闭锁、心脏缺陷、气管食管瘘、肾脏异常和肢体异常)综合征可能同时存在。最初的影像学表现可能正常,但随后的影像学检查通常显示进行性和复发性肺不张,这可能是由于通过异常气道反复误吸以及患侧肺在呼吸时顺应性差所致。在本报告中,我们描述了一名患有食管肺及罕见相关异常的新生儿,这些异常包括肛门直肠畸形、肺动脉吊带以及下腔静脉中断伴奇静脉延续。据我们所知,这是首次报道伴有此类关联的食管肺病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/3d76e6001a16/radcr449-fig-0004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/b20c7a162cb7/radcr449-fig-0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/e8fcef254886/radcr449-fig-0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/27cfc82fe825/radcr449-fig-0003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/3d76e6001a16/radcr449-fig-0004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/b20c7a162cb7/radcr449-fig-0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/e8fcef254886/radcr449-fig-0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/27cfc82fe825/radcr449-fig-0003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cd39/6000028/3d76e6001a16/radcr449-fig-0004.jpg

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本文引用的文献

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Communicating bronchopulmonary foregut malformation type IA: radiologic anatomy and clinical dilemmas.IA型交通性支气管肺前肠畸形:放射学解剖与临床困境
Surg Radiol Anat. 2015 Dec;37(10):1251-6. doi: 10.1007/s00276-015-1504-x. Epub 2015 Jun 16.
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Prenatal diagnosis of esophageal bronchus--first report of a rare foregut malformation in utero.食管支气管的产前诊断——子宫内一种罕见前肠畸形的首例报告
J Pediatr Surg. 2015 Feb;50(2):306-10. doi: 10.1016/j.jpedsurg.2014.11.025. Epub 2014 Nov 10.
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Diagnosis of esophageal lung combined with pulmonary sling using multidetector computed tomography.
为一名17个月大女童实施左区间电视胸腔镜肺切除术治疗II型交通性支气管肺前肠畸形。
Int J Surg Case Rep. 2020;77:235-242. doi: 10.1016/j.ijscr.2020.10.132. Epub 2020 Nov 2.
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Esophageal Lung in a Preterm Boy-Report on a Multidisciplinary Treatment.一名早产男婴的食管肺——多学科治疗报告
European J Pediatr Surg Rep. 2019 Jan;7(1):e110-e113. doi: 10.1055/s-0039-3400475. Epub 2019 Nov 28.
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Congenital bronchopulmonary foregut malformation: systematic review of the literature.先天性支气管肺前肠畸形:文献系统综述。
BMC Pediatr. 2019 Sep 2;19(1):305. doi: 10.1186/s12887-019-1686-1.
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Possible Approach to Esophageal Lung with Long Tracheobronchial Gap.治疗伴有长气管支气管间隙的食管源性肺的可能方法。
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Successful tracheobronchial reconstruction of communicating bronchopulmonary foregut malformation and long segment congenital tracheal stenosis: a case report.成功重建沟通性支气管肺前肠畸形和长段先天性气管狭窄的气管支气管:一例报告。
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