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日本呼吸器学会英文誌 Respiratory Investigation
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Abstract

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Article in Japanese

Case Report

A case of congenital tracheal stenosis in an adult

Tomohide Satoa  Chiharu Satoa  Mitsue Hayashib  Motohisa Yamasakib  Hiroshi Tukamotoc  Yuki Sumid 

aMutsuai Medical Clinic
bDepartment of Internal Medicine, Nitobe Memorial Nakano General Hospital
cDepartment of Radiology, Fujisawa City Hospital
dTokyo Medical and Dental University School of Health Sciences, Faculty of Medicine

ABSTRACT

A-23-year-old man visited our clinic with upper airway symptoms. Bilateral expiratory wheezes were detected on physical examination. The enlargement of the right pulmonary artery and lower bifurcation of the trachea observed on chest X-ray led us to perform chest computed tomography (CT). This revealed a narrowed trachea and left pulmonary artery malformation (pulmonary artery sling). A three-dimensional (3D) CT and a virtual bronchoscopy were performed on clinical suspicion of anatomical abnormality. The image showed concentric rings at the site of the tracheal narrowing (complete tracheal rings); we therefore made a diagnosis of congenital tracheal stenosis (CTS). Complete tracheal rings and pulmonary artery sling are typical findings in CTS. From 1980 to 2017, only 11 cases of CTS diagnosed in the adult phase were reported (comprising 9 females and 2 males, age at diagnosis 16 to 57 years). The median minimum tracheal diameter was 6.6mm, and no cases underwent tracheoplasty. Recently, cases diagnosed by 3D CT have been increasingly reported. Since abnormalities of the airway tract cause respiratory distress, it is extremely rare to survive the neonatal and infant phases without severe airway problems. Here, we report a case of CTS diagnosed in the adult phase and review the recent literature.

KEYWORDS

Congenital tracheal stenosis (CTS)  Complete tracheal ring  Pulmonary artery sling Virtual bronchoscopic navigation (VBN) 

Received 26 Sep 2017 / Accepted 1 Mar 2018

AJRS, 7(3): 182-186, 2018

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