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老年患者先天性囊性腺瘤样畸形合并支气管闭锁

Congenital cystic adenomatoid malformation with bronchial atresia in elderly patients.

作者信息

Kwak Hyun Jung, Moon Ji-Yong, Kim Sa-Il, Kim Tae Hyung, Sohn Jang Won, Kim Sang-Heon, Shin Dong Ho, Park Sung Soo, Chung Won Sang, Yoon Ho Joo

机构信息

Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea.

出版信息

Tuberc Respir Dis (Seoul). 2012 Jun;72(6):501-6. doi: 10.4046/trd.2012.72.6.501. Epub 2012 Jun 29.

DOI:10.4046/trd.2012.72.6.501
PMID:23101017
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3475458/
Abstract

UNLABELLED

Congenital cystic adenomatoid malformation (CCAM) is an uncommon, nonhereditary anomaly caused by arrest of lung. Patients with CCAM may present with respiratory distress as newborns, or may remain asymptomatic until later in life. CCAM type I is rarely found in association with bronchial atresia (BA) in adults; we present such a case.

CASE

A 54-year-old female presented with chronic cough and blood-tinged sputum. Physical examination and laboratory tests were unremarkable. Chest radiographs and a CT scan of the chest showed multiple large air-filled cysts consistent with a CCAM in the right lower lobe, and an oval-shaped opacity in the distal right middle lobal bronchus. Based on the radiologic findings, right middle lobectomy and a medial basal segmentectomy of the right lower lobe were performed via a thoracotomy. These lesions were consistent with Stocker's Type I CCAM and BA in the different lobes.

摘要

未标注

先天性囊性腺瘤样畸形(CCAM)是一种由肺发育停滞引起的罕见非遗传性异常。CCAM患者在新生儿期可能出现呼吸窘迫,也可能直到生命后期仍无症状。I型CCAM在成人中很少与支气管闭锁(BA)相关联;我们报告这样一例病例。

病例

一名54岁女性出现慢性咳嗽和痰中带血。体格检查和实验室检查无异常。胸部X线片和胸部CT扫描显示右肺下叶有多个与CCAM相符的大的含气囊肿,右肺中叶远端支气管有一个椭圆形致密影。根据影像学表现,通过开胸手术进行了右肺中叶切除术和右肺下叶内侧基底段切除术。这些病变在不同肺叶符合斯托克I型CCAM和BA。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/f4a1032c7c26/trd-72-501-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/02bc28b5063b/trd-72-501-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/4138935a077d/trd-72-501-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/3dbf2addb274/trd-72-501-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/f4a1032c7c26/trd-72-501-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/02bc28b5063b/trd-72-501-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/4138935a077d/trd-72-501-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/3dbf2addb274/trd-72-501-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9718/3475458/f4a1032c7c26/trd-72-501-g004.jpg

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本文引用的文献

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Congenital cystic adenomatoid malformation of the lung associated with bronchial atresia involving a different lobe in an adult patient: a case report.成人患者先天性肺囊性腺瘤样畸形合并支气管闭锁累及不同肺叶:一例报告
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Prenatal period to adolescence: the variable presentations of congenital cystic adenomatoid malformation.从孕期到青春期:先天性囊性腺瘤样畸形的多种表现形式
Pediatr Int. 2006 Dec;48(6):626-30. doi: 10.1111/j.1442-200X.2006.02264.x.
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Bronchial atresia is common to extralobar sequestration, intralobar sequestration, congenital cystic adenomatoid malformation, and lobar emphysema.支气管闭锁常见于肺叶外型肺隔离症、肺叶内型肺隔离症、先天性囊性腺瘤样畸形和大叶性肺气肿。
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Monaldi Arch Chest Dis. 2004 Jan-Mar;61(1):28-34.
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