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Ventilation-perfusion lung imaging appears to be a significant nonivasive means of supporting the clinical diagnosis of Swyer-James syndrome. Three cases are presented which display characteristic delayed washout of xenon from a small or normal-sized, underperfused hyperlucent lung.
Swyer-James syndrome (SJS) is usually diagnosed with plain chest radiographs obtained during inspiration/expiration. The authors studied patients with CT to assess its value in the evaluation of this syndrome. In patients with SJS, CT was useful in the determination of bronchial patency (all nine patients), lung parenchymal changes (subpleural infiltrates in six patients, atelectasis in two, and cavities in two), and the extent and degree of bronchiectasis (all nine patients). If CT is used for the evaluation of bronchiectasis, knowledge of the main findings associated with SJS (hyperlucent lung without anteroposterior gradient attenuation [n = 8], small lung [n = 6], and diminished central and peripheral pulmonary arteries [n = 9]) should facilitate the diagnosis of associated SJS. Expiration CT would support the diagnosis with demonstration of air trapping.
OBJECTIVE: Unilateral pulmonary artery agenesis (UPAA), a rare congenital anomaly that is frequently associated with other cardiovascular abnormalities, is usually diagnosed in childhood. Most patients who have no associated cardiac anomalies have only minor or absent symptoms and survive into adulthood. The conditions of such patients are frequently misdiagnosed in adulthood. In this report, we describe six patients with UPAA in whom the diagnosis was first established in adulthood. The varied clinical presentation of these patients is reviewed and the relative effectiveness of a variety of diagnostic tests is compared. SUBJECTS AND METHODS: During the period January 1987 through December 1990, six male patients, aged 17 to 20 years, were found to have UPAA at the time of their medical screening for enrollment into the armed forces. The diagnosis was based on history, clinical and imaging examinations, including chest radiography, ventilation-perfusion lung scan, digital subtraction angiography (DSA), computed tomography (CT), and magnetic resonance imaging (MRI). RESULTS: In four of the patients, the UPAA was on the left side and in two it was on the right. A right aortic arch was present in three patients and other cardiovascular anomalies were found in three. Pulmonary function studies showed a mild restrictive pattern in four. In contrast to previous reports, the ventilation scan showed a diminished "wash in" and "equilibrium" phase without a delayed "wash out" phase on the affected side in all patients. Selective bronchography through the fiberoptic bronchoscope revealed ipsilateral mixed-type bronchiectasis in two of four patients studied, a finding of clinical significance that has not been described previously. In all cases, the diagnosis was made by DSA. CT of the thorax (n = 6) and MRI (n = 4) were diagnostic in all cases in which they were performed, but added no significant information. CONCLUSION: UPAA is frequently misdiagnosed in adulthood and is often not considered in the differential diagnosis of the unilateral hyperlucent lung. Clinicians and radiologists should be aware of the possibility of undiagnosed cases in adults, with many atypical characteristics.
A case of hyperlucent lung, the Swyer-James syndrome, diagnosed by computed tomography (CT) scan is presented. The CT findings and the role of this modality in establishing the diagnosis are discussed.
Isolated unilateral pulmonary artery agenesis is a rare condition, which in most patients is asymptomatic. Occasionally patients present with symptoms that are nonspecific and not necessarily attributable to disease of the respiratory system. In these individuals the clue to the diagnosis is found in a plain chest roentgenogram, often revealing a hyperlucent contracted hemithorax. We present an unusual case of isolated unilateral pulmonary artery agenesis associated with the opportunistic organism Mycobacterium kansasii and Aspergillus fumigatus in which the diagnosis was made 10 years after initial presentation. Clinicians should be aware of this condition and include it in their differential diagnosis of a hyperlucent lung field on the chest roentgenogram.
Bacterial (including intracellular pathogens) and mainly viral pneumonia can lead to middle and long term pulmonary sequelae in children with or without underlying disease, namely chronic atelectasis (right middle lobe syndrome), bronchiectasis, obliterative bronchiolitis, unilateral hyperlucent lung syndrome, fibrosis, and peumatoceles. Functional alterations may also be observed, such as bronchial hyperreactivity, chronic cough and asthma. Additionally a relationship between pneumonia in early childhood and the further occurrence of chronic obstructive pulmonary disease in adult life is now admitted. The occurrence of sequelae being usually unpredictable, and a careful assessment of the clinical and radiological outcome is important. The unusual persistence of either respiratory symptoms or radiological alterations justifies investigations such as tomodensitometry and the evaluation of lung function, to look for complications or underlying diseases and, whenever it is possible, to set up an adapted treatment.
Adenoviruses are well known causes of respiratory illness in children. Long term sequelae reported with types 3, 7, and 21 include bronchiolitis obliterans, bronchiectasis, and the hyperlucent lung or McLeod syndrome. Twenty children admitted to hospital with adenovirus type 7 pneumonia between 1960 and 1978 were studied and compared with 20 controls admitted during the same period with adenovirus type 7 upper respiratory tract infections. Sixty five per cent of the pneumonia group had developed evidence of airways obstruction compared with 10% of controls. Young age at the time of pneumonia and a 'measles-like' illness before its onset increase the chance of developing long term pulmonary function abnormalities. Sex and family history of smoking or atopy do not influence outcome.
A 15 year old girl who was labelled as having MacLeod's syndrome on the basis of a chest radiograph was subsequently, at the age of 23 years, found to have an adenoid cystic carcinoma obstructing the left main bronchus. This case emphasises the need to exclude a central lesion in cases of unilateral hyperlucent lung.
OBJECTIVE: The functional consequences of pulmonary emphysema have been estimated from autopsy studies. This antemortem study correlates structure as assessed by criteria applied to chest radiographs and function in earlier disease. SUBJECTS AND METHODS: Four thousand one hundred seven male shipyard and construction workers had chest radiographs scored for asbestosis using International Labour Office criteria and for emphysema using five criteria: hyperlucent lung fields, low flat diaphragms on posteroanterior views, low flat diaphragms on lateral radiographs, a wide retrosternal space, and bullae. Total lung capacity (TLC) was measured by planimetry of radiographs and forced vital capacity (FVC) and flows by spirometry: TLC minus FVC equaled residual volume (RV). All were expressed as percentage of predicted (% pred) adjusted for height, age, and duration of cigarette smoking. Linear regression modeling examined the influence of each emphysema criterion on function measurements. RESULTS: Four hundred three (10.1%) of 4,107 men had two or more emphysema criteria and 24.5% had asbestosis. Prevalence of 2 criteria varied by smoking status and was present in 20% of current smokers, 7.5% of ex-smokers, and 2% of never smokers. The TLC % pred and RV/TLC increased and % pred of FEV1/FVC and FEF25-75, FEV1, and FVC decreased with increasing radiographic emphysema criteria. Forty-four (10%) had normal results of spirometry with two or more radiographic emphysema criteria. CONCLUSION: Impairment of airway function worsened with each additional radiographic criteria of emphysema. These criteria were more strongly associated with an increase in RV/TLC than an increase in TLC.
OBJECTIVE: The aim of the study was to determine the clinical profile, aetiology and radiological categories in children diagnosed with bronchiolitis obliterans (BO). METHODOLOGY: We undertook a review of the medical records and radiological studies of 14 children with BO. RESULTS: Unresolving cough and wheeze after a short respiratory illness was the commonest presentation. A viral pneumonitis was identified as the initial respiratory event prior to the development of BO in six children and Mycoplasma pneumoniae was the cause in another three children. Chest X-ray findings could be divided into four distinct patterns that were hyperinflation (n=5), mixed pattern of atelectasis, hyperlucency and bronchial thickening (n=4), unilateral small hyperlucent lung (n=3) and unilateral collapse of one lung (n=2). High resolution computed tomogram (HRCT) chest showing areas of hyperaeration and mosaic ground glass patterns with bronchial thickening were commonly found in patients whose chest X-ray showed bilateral changes. Patients with bilateral lung changes were more likely to have failure to thrive and persistent respiratory symptoms on follow up. CONCLUSION: A diagnosis of BO can be made from typical clinical features combined with an understanding of the different chest X-ray categories and HRCT of the chest. A viral aetiology was the commonest cause for BO in our series.
Congenital bronchial atresia (CBA) is a congenital anomaly but is seldom diagnosed in children. They are usually asymptomatic and are found during screening chest radiographs in young adults. The characteristic chest radiographs of CBA consist of a hyperlucent lung, central branching but contrast non-enhancing mass and air-fluid lesion. Most of the cases reported in the past were managed surgically. The present study reported an 11-yr-old presenting with chest pains, exercise intolerance, and refractory wheezing. It is of utmost importance to consider the possibility of CBA in children with lobar emphysema in order to achieve early diagnosis and avoid unnecessary exploratory surgery.
Lesions that cause decreased pulmonary blood flow are those that obstruct the RVOT. In general, the radiographic presentation includes the following: (1) Thin, poorly visualized pulmonary blood vessels with little extension beyond the perihilar region; (2) Dark, hyperlucent lung fields; and (3) Moderate to severe cardiomegaly, depending on the cardiac defect(s) present.
The Mac Leod syndrome is a unilateral pulmonary hypoplasia with emphysema. Onset usually occurs in childhood. The authors delineate this well-defined nosologic entity which is one of the many etiologies of the unilateral hyperlucent lung.
Two cases of total occlusion of the right pulmonary artery 22 and 28 years after mediastinal irradiation are presented. The first patient is alive 29 years after the radiation therapy, and the second patient died of heart failure 31 years after the therapy. This very rare complication is one cause of radiological unilateral hyperlucent lung syndrome. The diagnosis can be confirmed by clinical history, clinical examination, chest roentgenogram, lung scan and pulmonary angiography.
The paper reports a case of silicosis associated with idiopathic unilateral hyperlucent lung--MacLeod's disease (ML)--in a 65-year old miner. ML had not been diagnosed before this clinical observation and the patient had been exposed to silica dust for about 15 years as a truck operator in water supply tunnelling work in central Italy. Impaired ventilatory function due to left pulmonary dystrophy was the favouring or even the causal factor of dust accumulation in the functionally active right lung and of the tissue reaction to the dust, leading to interstitial fibrosis. A radiological diagnosis of p 1/0 pneumoconiosis was made according to the 1980 International Labour Office classification. Clinical, radiological and functional findings indicated the presence of respiratory failure as a result of the sum of silicotic and dystrophic lesions. Two combined pulmonary scintigraphic examinations with Tc99 perfusional and mTc-DPTA inhalatory methods revealed peculiar alterations: absence of ventilation associated with severe vascular impairment in the left lung, inequality of the ventilation/perfusion rate, due to pneumoconiosis, in the right lung. MacLeod's disease is rare, but must be considered as a mandatory contra-indication for work involving dust exposure risk since it is one of the predisposing factors which can cause an occupational disease even if environmental exposure standards are observed.
Swyer-James syndrome is considered to be a relatively uncommon disease entity presenting with unilateral hyperlucent lung due to hypoplasia of a pulmonary artery and bronchiectasis of the affected lung. Association of bronchogenic carcinoma with Swyer-James syndrome has not apparently been reported in any previous literature, except for one Japanese case. In the present paper, we describe a 48-year-old male individual, who developed poorly differentiated squamous cell carcinoma of the right upper lobe bronchus after he had been diagnosed to have Swyer-James syndrome with unilateral hyperradiancy of the left lung. It is suggested that the bronchial epithelium of the unaffected lung in Swyer-James syndrome is likely to be more exposed to extrinsic carcinogens than that of the affected, underventilated lung, hence resulting in a higher risk of developing bronchogenic carcinoma.
Swyer-James syndrome, unilateral hyperlucent lung with air entrapment, generally occurs after severe infections during childhood. It is usually diagnosed by its characteristic chest radiographic image film or computed tomography, in patients who are almost asymptomatic. We report a case of Swyer-James syndrome, diagnosed from the study of severe pulmonary hypertension and with a fatal outcome.
Macleod/Swyer-James syndrome is an uncommon and complex disease characterized by roentgenographic hyperlucency of one lung or lobe due to loss of the pulmonary vascular structure and to alveolar overdistension. This syndrome seems to be an acquired disease that follows viral bronchiolitis and pneumonitis in childhood. It must be differentiated from many other causes of unilateral lung "transradiancy" on the chest roentgenogram, such as those related to congenital bronchial and/or vascular abnormalities. We here describe an 11-year-old patient with Macleod/Swyer-James syndrome and bronchiectasis resulting in severe recurrent bronchopulmonary infections. Despite the severe impairment of pulmonary function, the patient underwent resection of the right lung with progressive improvement of clinical and physiologic parameters.