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Swyer-James syndrome associated with asthma and a giant bulla.

Kayawake H, Sakai H, Matsumoto H, Date H - Respirol Case Rep (2014)

Bottom Line: The characteristic feature of this syndrome is unilateral pulmonary hyperlucency on a chest X-ray film.Typical symptoms include recurrent chest infections, chronic cough, wheezing, and exertional dyspnea.Although there have been a few reports of pneumothorax in patients with Swyer-James syndrome, there have been no reports about an association with giant bullae.

View Article: PubMed Central - PubMed

Affiliation: Department of Thoracic Surgery, Kurashiki Central Hospital Okayama, Japan ; Department of Thoracic Surgery, Kyoto University Kyoto, Japan.

ABSTRACT
Swyer-James syndrome was first described in 1953 as unilateral pulmonary emphysema in a 6-year-old boy. The characteristic feature of this syndrome is unilateral pulmonary hyperlucency on a chest X-ray film. Typical symptoms include recurrent chest infections, chronic cough, wheezing, and exertional dyspnea. Although there have been a few reports of pneumothorax in patients with Swyer-James syndrome, there have been no reports about an association with giant bullae. Here, we report a case of Swyer-James syndrome associated with a giant bulla and asthma, in which surgery achieved marked improvement of dyspnea.

No MeSH data available.


Related in: MedlinePlus

Chest X-ray films and chest computed tomography scans show a giant bulla in the right upper lobe, which enlarges over time. Hyperlucency in the right lower lobe is caused by diminished vasculature because of a hypoplastic right pulmonary artery. There is no evidence of bronchiectasis.
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fig01: Chest X-ray films and chest computed tomography scans show a giant bulla in the right upper lobe, which enlarges over time. Hyperlucency in the right lower lobe is caused by diminished vasculature because of a hypoplastic right pulmonary artery. There is no evidence of bronchiectasis.

Mentions: Based on the detection of unilateral pulmonary hyperlucency and the absence of other possible causes, including congenital cysts, congenital pulmonary artery abnormalities, and embolization of the main pulmonary artery, she was diagnosed as having Swyer-James syndrome with a giant bulla. In addition, her response to an inhaled bronchodilator, the diurnal variation of PEF, and the detection of eosinophilic airway inflammation suggested the recurrence of her asthma. Although surgical resection of the giant bulla was considered, medical treatment of the patient's asthma was given precedence to maximize her respiratory function. After treatment with an inhaled corticosteroid preparation and tulobuterol patch, her exertional dyspnea showed marked improvement, and the exhaled nitric oxide level and PEF also improved. From August 2009, however, she complained of right-sided chest pain and her exertional dyspnea recurred. Simultaneously, the giant bulla in the right upper lobe enlarged further and the patient's inspiratory capacity decreased (Figure 1). Because her chest pain and exertional dyspnea continued to worsen despite higher doses of asthma medication, surgical resection of the giant bulla was performed in January 2011.


Swyer-James syndrome associated with asthma and a giant bulla.

Kayawake H, Sakai H, Matsumoto H, Date H - Respirol Case Rep (2014)

Chest X-ray films and chest computed tomography scans show a giant bulla in the right upper lobe, which enlarges over time. Hyperlucency in the right lower lobe is caused by diminished vasculature because of a hypoplastic right pulmonary artery. There is no evidence of bronchiectasis.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4263499&req=5

fig01: Chest X-ray films and chest computed tomography scans show a giant bulla in the right upper lobe, which enlarges over time. Hyperlucency in the right lower lobe is caused by diminished vasculature because of a hypoplastic right pulmonary artery. There is no evidence of bronchiectasis.
Mentions: Based on the detection of unilateral pulmonary hyperlucency and the absence of other possible causes, including congenital cysts, congenital pulmonary artery abnormalities, and embolization of the main pulmonary artery, she was diagnosed as having Swyer-James syndrome with a giant bulla. In addition, her response to an inhaled bronchodilator, the diurnal variation of PEF, and the detection of eosinophilic airway inflammation suggested the recurrence of her asthma. Although surgical resection of the giant bulla was considered, medical treatment of the patient's asthma was given precedence to maximize her respiratory function. After treatment with an inhaled corticosteroid preparation and tulobuterol patch, her exertional dyspnea showed marked improvement, and the exhaled nitric oxide level and PEF also improved. From August 2009, however, she complained of right-sided chest pain and her exertional dyspnea recurred. Simultaneously, the giant bulla in the right upper lobe enlarged further and the patient's inspiratory capacity decreased (Figure 1). Because her chest pain and exertional dyspnea continued to worsen despite higher doses of asthma medication, surgical resection of the giant bulla was performed in January 2011.

Bottom Line: The characteristic feature of this syndrome is unilateral pulmonary hyperlucency on a chest X-ray film.Typical symptoms include recurrent chest infections, chronic cough, wheezing, and exertional dyspnea.Although there have been a few reports of pneumothorax in patients with Swyer-James syndrome, there have been no reports about an association with giant bullae.

View Article: PubMed Central - PubMed

Affiliation: Department of Thoracic Surgery, Kurashiki Central Hospital Okayama, Japan ; Department of Thoracic Surgery, Kyoto University Kyoto, Japan.

ABSTRACT
Swyer-James syndrome was first described in 1953 as unilateral pulmonary emphysema in a 6-year-old boy. The characteristic feature of this syndrome is unilateral pulmonary hyperlucency on a chest X-ray film. Typical symptoms include recurrent chest infections, chronic cough, wheezing, and exertional dyspnea. Although there have been a few reports of pneumothorax in patients with Swyer-James syndrome, there have been no reports about an association with giant bullae. Here, we report a case of Swyer-James syndrome associated with a giant bulla and asthma, in which surgery achieved marked improvement of dyspnea.

No MeSH data available.


Related in: MedlinePlus