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PubMed1993

[Two cases of Swyer-James syndrome].

Case 1: 55-year-old male was admitted to our hospital for further examination of increased transparency of X-ray in the left lower lung. He had history of pneumonia in his childhood. Left bronchography revealed mild cylindrical bronchi-ectasia in the proximal bronchi but poor filling by contrast in the peripheral bronchi. Case 2: 61-year-old male was referred to our hospital with palpitation and dyspnea. Chest X-ray film revealed hyperlucency of the left lower lung. The ventilation scan showed a marked decrease in ventilation to the left lung and air trapping was present in the left lung. The perfusion scan showed a matched decrease in blood supply to the left lung. In both cases, pulmonary arteriogram showed narrowed and withered-tree-like left pulmonary arteries. From these clinical findings, we diagnosed these two cases as Swyer-James syndrome. We are reporting two cases of Swyer-James syndrome and describing the clinical feature, differential diagnosis and etiology of the syndrome.

Arteriosclerosis↗
PubMed2001

Congenital cystic adenomatoid malformation type 4.

A 9-day-old boy presented in respiratory distress and with failure to thrive. The chest X-ray showed a hyperlucent area of the left lung. A resection of the markedly emphysematous segment 2 of the left upper lobe was performed assuming the emphysematous tissue was due to congenital lobar emphysema (CLE). Histological examination of the lung tissue, however, revealed a pattern consistent with congenital cystic adenomatoid malformation (CCAM) type 4. The therapy for CLE as well as for CCAM is similar, i.e., resection of the emphysematous tissue. As far as the prognosis is concerned, it is important to diagnose the exact type of malformation in order to exclude associated anomalies, as well as the risk of development of malignancies in later life. The frequency of associated malformations of CCAM type 4 is unknown. Although the risk for development of malignancies from CCAM type 4 is not clear at the moment, the possible development of malignancies justifies prompt resection shortly after diagnosis, even in asymptomatic patients. A life-long follow-up in those patients who had a resection of CCAM in early childhood is recommended.

Cystic Adenomatoid Malformation of Lung, Congenita↗
PubMed1994

[A case of surgical treatment of Swyer-James syndrome].

Swyer-James syndrome was usually diagnosed on the past history and the radiologic features, and almost all the cases were followed by the conservative therapy. Therefore, the examination of the pathologic features was rare. We recently experienced a patient with Swyer-James syndrome, who had a history of respiratory infection from his childhood onward and whose chest radiograph revealed hyperlucency of the right lower lung field. A standard right middle and lower lobectomy was performed. Histological examination of the resected specimens revealed hypoplasia of alveoli and pulmonary artery.

Adult↗
PubMed1980

Crippled lung: variations on a theme by Macleod.

We have studied nine male patients (age 18 to 68 years) with radiographic and physiologic evidence of an abnormally small lung on one side (three right and six left). All had had a childhood pneumonia or bronchiolitis and eight had chronic or recurrent bronchitis and exertional dyspnea. Radiography showed two of the small lungs to be hypolucent while seven were hyperlucent. Bronchography revealed evidence of bilateral chronic bronchitis in all with saccular bronchiectasis in three. Angiography showed strikingly diminished vascularity to the smaller lung. Spirometry revealed airway obstruction in seven of the patients. All had pulmonary arterial hypertension. Radiospirometry showed that the small lung had on the average 30% of the total ventilation but only 15% of the perfusion. Washout of 133 Xe was extremely slow in radiolucent regions. We suggest the name "crippled lung" syndrome for this entity because it is purely descriptive and encompasses several clinical variants. It also avoids the pitfalls of etiologic implication (acquired-congenital). Clinical or subclinical bronchitis seems to be common in these patients and the prime goal in therapy must be to combat the tendency towards airway infection.

Adolescent↗
PubMed1992

Swyer-James syndrome: CT findings in eight patients.

To determine the importance of chest CT findings in patients with Swyer-James syndrome (unilateral small lung with air trapping) and to compare these findings with those on chest radiographs and scintigrams, we reviewed the CT scans, chest radiographs, and scintigrams of eight patients with the syndrome. Radiographs showed unilateral hyperlucency in seven patients and bilateral asymmetric hyperlucency in one. CT showed that the hyperlucency was unilateral in only three and that hyperlucency in one. CT showed that the hyperlucency was unilateral in only three and that hyperlucent regions on radiographs contained patches of normal lung attenuation in five patients. Conversely, in four patients, CT also showed small hyperlucencies in regions considered normal on radiographs. These lucencies usually had poorly defined margins and irregular shapes (five patients), but sometimes were peripheral, wedge shaped, and sharply demarcated (two patients). CT also showed subtle abnormalities not visible on radionuclide scans in two patients. Air trapping in hyperlucent regions was confirmed by a lack of change in volume on expiratory CT scans in five cases. Bronchiectasis was found in only three patients. CT helps to exclude central bronchial obstruction, cysts, and vascular disease as causes of hyperlucency. By excluding central obstruction, CT may make bronchoscopy unnecessary in some patients. CT is more sensitive than radiographs and radionuclide scans in detecting hyperlucent regions and in showing their distribution. Our experience suggests that bronchiectasis is not a necessary component of the Swyer-James syndrome.

Adolescent↗
PubMed1998

[Congenital bronchial atresia complicated with severe cough--a case report].

A 37-year-old woman presented severe cough for several years. Chest X-ray showed an abnormal shadow in the right upper lung field. That was absent but hyperlucent before onset. Chest CT scan revealed a mass shadow with mucoid impaction in the right S2. Thoracoscopic right upper lobectomy dramatically improved her complaints. Histological examination revealed the dilated bronchus containing mucoid impaction and confirmed a diagnosis of congenital bronchial atresia. It was suggested that organizing pneumonia resulting from repeated infection caused severe cough. Thoracoscopic surgery for congenital bronchial atresia should be recommended in young patients.

Adult↗
PubMed1977

Congenital lobar emphysema.

Congenital lobar emphysema is frequently a life-threatening disorder presenting in infancy. The diagnosis should be considered when the patient shows signs of respiratory distress and the x-ray demonstrates the characteristic hyperlucent lobe with compression of the surrounding lung tissue. The morphologic aspects and pathogenesis are not clearly defined, although it is thought that an abnormality of bronchial cartilage is probably associated with the development of lobar emphysema. The results of treatment by lobectomy are excellent, and the mortality with surgery is low. Consideration of nonsurgical management is worthwhile in mildly affected or asymptomatic patients. Long-term follow-up of both groups indicates a very favorable prognosis in this disease entity.

Humans↗
PubMed1991

[A case of Swyer-James syndrome diagnosed by selective alveolobronchography].

A 33-year-old male was admitted to our hospital for further investigation of unilateral hyperlucency of the left upper and middle lung fields on chest X-ray. A pulmonary scan showed decreased perfusion of the left upper and middle lung fields. Pulmonary arteriography demonstrated small left pulmonary arteries with diminished peripheral vascular branches. Left bronchogram showed almost normal distribution of main and subsegmental bronchi, but poor filling by contrast in the peripheral bronchi. From these findings, the patient was diagnosed as having Swyer-James syndrome. In order to demonstrate the alveoli in this syndrome, selective alveolobronchography (SAB) was performed. SAB revealed coarse ring shadows of B1+2.

Adult↗
PubMed2000

Chest radiograph heterogeneity predicts functional improvement with volume reduction surgery.

BACKGROUND: Using a historical cohort study model, we tested the hypothesis that heterogeneity of emphysematous changes on the preoperative chest radiograph correlated with favorable outcome of lung volume reduction surgery. METHODS: The test population consisted of 21 patients with severe emphysema who were being treated at a 1,000-bed university-affiliated tertiary teaching hospital. A simple but quantitative index of heterogeneity has been devised, whereby the preoperative posteroanterior chest radiographic lung fields are divided into four geometric quadrants. Each quadrant is scored (0 to 4) for emphysematous changes by two radiologists blinded as to subsequent patient management and outcome. Criteria for determining presence of emphysema were hyperlucency, decreased vascular markings, and parenchymal crowding indicating compressed lung. Heterogeneity index is the sum of the two highest scores minus the two lowest, with a maximum index of 8 and a minimum of 0. Preoperative chest radiographs and postoperative changes in forced expiratory volume in 1 second were examined. RESULTS: The heterogeneity index was positively correlated with change in forced expiratory volume in 1 second after operation with an r2 of 0.31 and an average increase of 117 mL per unit increase in heterogeneity index (p < 0.009). CONCLUSIONS: This simple index of heterogeneity may be useful as a predictor of improved pulmonary function after lung volume reduction surgery.

Aged↗
PubMed1985

Pulmonary air cysts in cystic fibrosis: relation of pathologic features to radiologic findings and history of pneumothorax.

One lung obtained from each of 21 consecutive autopsies in adolescents and young adults with cystic fibrosis was studied prospectively by macroscopic morphometry and light microscopy to determine the prevalence, morphology, and radiographic appearance of subpleural air cysts, which potentially contribute to spontaneous pneumothorax. In 15 lungs, 41 cysts of three anatomic types were identified: bronchiectatic cysts (23), interstitial cysts (13), and emphysematous bullae (5). All cysts were significantly more numerous in the upper lobe. Bronchiectatic cysts had the largest mean diameter, occupied from less than 1 per cent to 47.7 per cent of upper lobe volume in nine patients, and produced large multiloculated hyperlucencies on chest radiographs in five cases. All six lungs with prior pneumothorax contained at least one cyst, but no significant difference was found in the type or proportion of lung volume occupied by cysts between lungs with and without pneumothorax. Patients with large cysts had significantly lower chest radiograph scores, but there was no correlation between the proportion of lung volume occupied by cysts and patient age or duration of either symptomatic lung disease or colonization by bacteria. On chest radiographs only bronchiectatic cysts with conglomerate diameters of greater than 3 cm were visible. Smaller lesions could not be separated from ring shadows produced by bronchiectasis.

Adult↗
PubMed1987

[Characteristics of respiratory functional involvement in MacLeod's syndrome (or Swyer-James syndrome)].

MacLeod's syndrome (Swyer-James' syndrome in the child) is a radiologic entity: hyperlucency of one or several lobes, or even of one lung, scarce hilar shadow on the same side as the abnormal transradiency, and barely visible arterial network on the abnormal side of the thorax. Distal bronchiectases (bronchography) and abnormal distal bronchi (necropsy) suggest that this disease may be due to bronchiolitis in childhood. An obstructive pulmonary disorder, assessed by pulmonary function tests, has been described in this syndrome. However, neither the prevalence nor the severity or evolution of the obstructive defect are known. We have studied two such patients for 5 and 12 years respectively: during this long follow-up period the obstructive disorder, albeit severe, remained stable. We have also analysed the 75 cases in the literature in which pulmonary function tests are available and comparable. We have been able to show that (1) an obstructive disorder is frequent in MacLeod's syndrome, (2) the severity of this disorder differs markedly between patients, and (3) this airflow limitation is probably stable on a long-term basis. These findings are compatible with the pathogenetic hypothesis at present accepted, ie bronchiolitis during childhood, and suggest that the inflammatory bronchial disease may have been generalized in many such patients.

Adult↗
PubMed1981

Supine subpulmonary pneumothorax.

Intrapleural air often assumes a subpulmonary position in the recumbent patient even in the absence of underlying pulmonary disease. The margins of the collapsed lung may be difficult or impossible to detect, and a poorly defined basilar hyperlucency may be the only clue present. Additional helpful findings include depression and clear visualization of the diaphragm anteriorly (anterior costophrenic sulcus) creating a "double" appearance to the diaphragm, an unusually distinct cardiac apex, and pericardial fat tags. These signs should help alert the viewer to the correct diagnosis of pneumothorax.

Diaphragm↗
PubMed1997

Disappearing fetal lung masses: importance of postnatal imaging studies.

BACKGROUND: The "disappearance" of congenital masses of the lung on prenatal sonograms has been described, but the importance of postnatal imaging studies in these children is unknown. OBJECTIVE: The objective of this work was to study the utility of radiographs and CT scans in asymptomatic infants with congenital masses of the lung that partially or completely resolve on prenatal sonograms performed late in gestation. MATERIALS AND METHODS: The prenatal sonograms, postnatal imaging studies, surgical findings, and pathologic diagnoses of seven children with an echogenic mass of the lung that improved or disappeared on prenatal sonograms were reviewed. RESULTS: All masses were type II congenital cystic adenomatoid malformation, with features of intralobar sequestration also being found in four. An unsuspected extralobar sequestration adjacent to a left lower lobe mass was found at surgery in one patient. All masses were hyperechoic compared with normal lung on sonograms prior to 32 weeks of gestation, with cysts being seen in four. On scans after 32 weeks, four of the masses had resolved completely and three showed subtle increased echogenicity compared with normal lung. Cysts completely resolved in two of four cases. Postnatal radiographs showed subtle abnormalities in four infants, a hyperlucent lobe in one, a soft tissue mass with adjacent hyperlucency in one, and normal findings in one. CT scans were abnormal in all cases, with air-filled cysts and soft tissue in six and a hyperinflated lobe in one. CONCLUSION: Children with "disappearing" fetal lung masses have persistent abnormalities after birth that are often subtle on radiographs but are well demonstrated with CT.

Cystic Adenomatoid Malformation of Lung, Congenita↗
PubMed1994

Unilateral hyperlucent thorax on plain chest radiographs after neck dissection: importance of atrophy of the trapezius muscle.

OBJECTIVE: A hyperlucent thorax on plain chest radiography indicates a decrease in the radiographic density of the thorax, which can be caused by intra- or extrapulmonary diseases. The purpose of this study was to assess the prevalence and mechanisms that may be responsible for unilateral hyperlucency of the thorax after neck dissection and to determine if atrophy of the trapezius due to the transection of the accessory nerve is a cause of hyperlucent thorax. MATERIALS AND METHODS: Differences in the radiographic density between the right and left lung were evaluated and correlated with transection of the accessory nerve in 21 patients who had had a radical or a modified neck dissection for a malignant tumor of the head and neck. Twenty-eight neck dissections were performed on these 21 patients (seven had a simultaneous bilateral neck dissection). In 14 of the 21 patients, the accessory nerve had been severed during the neck surgery. In six patients, mechanisms responsible for a hyperlucent thorax were investigated with follow-up thoracic CT scans. RESULTS: In the radiographs, eight patients had a hyperlucent thorax on the side of the neck dissection. In all of these cases, the accessory nerve on the side of this neck dissection had been severed during a radical or a modified neck dissection. Prior to surgery, no such hyperlucency was noted. CT scans showed atrophy of the denervated trapezius muscle. CONCLUSION: Our findings show that atrophy of the denervated trapezius muscle after neck dissection is a cause of unilateral hyperlucent thorax on plain chest radiographs. Therefore, this finding should be anticipated as a postoperative change in patients who have had this surgery.

Accessory Nerve↗
PubMed1999

Pediatric diffuse lung disease: diagnosis and classification using high-resolution CT.

OBJECTIVE: Our purpose was to categorize high-resolution CT findings in children with diffuse lung disease and to evaluate the accuracy of diagnoses made using CT. MATERIALS AND METHODS: The chest radiographs and high-resolution CT scans of 20 children (1-16 years old; median, 9 years old) with biopsy-proven chronic diffuse lung diseases were reviewed separately by two independent chest radiologists. Thirteen types of diffuse lung disease were included in the study. Radiographic and CT features were noted, and three choices of diagnosis were recorded, with the confidence level. RESULTS: Diagnoses were made with a high degree of confidence (definite or probable) in 25 of 40 interpretations of CT scans, compared with only five of 40 interpretations of chest radiographs (p < .001). Fourteen (56%) of the 25 confident first-choice diagnoses on CT scans were correct, compared with two (40%) of the five interpretations on chest radiographs. Diseases were classified as belonging to one of five distinct groups on the basis of dominant CT features. Airway disease (n = 5) (bronchiolitis obliterans or bronchocentric granulomatosis) showed geographic hyperlucency on CT. Septal disease (n = 4) (lymphangiomatosis, hemangiomatosis, or microlithiasis) showed septal thickening. Infiltrative lung disease (n = 7) (desquamative interstitial pneumonitis, hypersensitivity pneumonitis, or lymphoid interstitial pneumonitis) showed ground-glass opacity. Air-space disease (n = 3) (aspiration, vasculitis, or bronchiolitis obliterans organizing pneumonia) showed lung consolidation. Langerhans' histiocytosis (n = 1) showed cysts and nodules. Surprisingly little overlap was seen among these groups. CONCLUSION: CT increases the level of diagnostic confidence for pediatric infiltrative lung disease, improves diagnostic accuracy, and provides a useful classification system.

Child↗
PubMed1990

[Diagnosis of bullae and blebs using contrast pleurography].

Twenty-five patients with spontaneous pneumothorax were studied with contrast pleurography. The technique was referred to that of rudy introduced. It could clearly show not only the shape of pleural surfaces and the anatomic relations of interlobular spaces, parietal and visceral pleurae, mediastinal and diaphragmatic boundaries, but also the signs of bullae and blebs, while the contrast agent flowed in pleural cavity. The diagnostic rate on bulla and bleb was 100%, which was superior to that of the plain roentgenogram. The big or small hyperlucent vesicular inflated lesions, most were multiple and got together, were observed in the lung with pneumothorax. Thoracotomies were performed in 4 cases and the locations of bullae were found to accord with the results of pleurography. Side-effect was not observed during or after this procedure.

Adult↗
PubMed1997

High-resolution CT in long-term follow-up after lung transplantation.

OBJECTIVE: Our aim was to evaluate the development of changes on high-resolution CT (HRCT) associated with chronic pulmonary rejection. MATERIALS AND METHODS: Repeated HRCT examinations were performed 140 times on 13 consecutive lung transplant recipients during a mean observation period of 26 months. The postoperative time interval to the first detection of each chronic change on CT was calculated and compared with the onset of chronic rejection. Bronchiolitis obliterans syndrome (BOS) or the histologic diagnosis of obliterative bronchiolitis was assessed by the published criteria of the International Society for Heart and Lung Transplantation. RESULTS: BOS developed in eight patients, on an average, within 11.6 (+/-5.0) months. Histologic diagnosis was available from five patients. On HRCT, among the first identifiable chronic changes were volume contraction, decreased peripheral vascular and bronchial markings, and thickening of septal lines, all of which appeared between 7 (+/-5.0) and 11 (+/-6.8) months postoperatively. The mean interval for appearance of bronchodilatation was 12.5 (+/-8.7) months. Hyperlucency and mosaic phenomenon were identified, on an average, 16 (+/-6.3) and 21 (+/-7.3) months after transplantation. CONCLUSION: On radiologic monitoring of lung recipients with HRCT, in addition to bronchodilatation. a special attention should be paid to the early chronic changes, including diminution of peripheral bronchovascular markings, thickening of septal lines, and volume reduction, which usually precede the establishment of the diagnosis of chronic rejection, whereas hyperlucency and mosaic phenomenon usually appear during more advanced BOS.

Bronchiolitis Obliterans↗