Chronic hypersensitivity pneumonitis due to isocyanate in a patient presenting with acute symptoms 1 month after environmental exposure

A 51-year-old man who had been working for 10 years with polyurethane paint containing isocyanate (MDI) was admitted to our hospital with complaints of fever and exertional dyspnea. Fine crackles were heard in both bases, and the patient had clubbed fingers. A chest X-ray film and computed tomograms of the lungs revealed patchy infiltrative shadows in both lung fields and subpleural honeycombing associated with irregular linear areas. Examination of bronchoalveolar lavage fluid showed increased T lymphocytes and a decreased CD 4/8 ratio. Specimens obtained by transbronchial lung biopsy revealed lymphoplasmacytic infiltration into the thickened alveolar walls, macrophage accumulation, and micro-epithelioid cell granulomas in the alveolar sacs. Hypersensitivity pneumonitis was suspected although the causative antigen was not identified because the results of short-term environmental provocation tests were negative in the patient's home and workplace. After discharge, the patient continued working as a paint sprayer. His acute symptoms recurred 1 month after exposure to isocyanate. Similar episodes occurred on two separate occasions. In addition, the patient tested positive for antibody to MDI-HSA in bronchoalveolar fluid. From the above observations, the patient was given a diagnosis of chronic hypersensitivity pneumonitis due to isocyanate (MDI). This condition is extremely rare. Furthermore, it is interesting that acute symptoms recurred 1 month after environmental exposure to the causative antigen.     

Childhood hypersensitivity pneumonitis due to dove antigens

A case of hypersensitivity pneumonitis due to doves is reported and compared with other cases due to dove or pigeon antigens reported in children. The diagnosis is substantiated by the presence of precipitating antibody to dove and pigeon serum, clinical improvement after contact with the doves was broken, and a positive response to inhalation challenge with pigeon serum. The insidious nature of this disease is emphasized as well as the importance of having detailed environmental information in children with unexplained respiratory disease.     

Matchmakers'' "phossy jaw" eradicated

At the University of Maryland Medical Systems, 356 consecutive thoracoscopic procedures were performed including 147 lung resections for various indications. Forty-nine patients underwent thoracoscopy for the diagnosis of interstitial lung disease. Two patients underwent bilateral procedures after a gap of more than six months for suspected malignancy. There were 28 females and 21 males. Age ranged from 23 to 75 years. The mean length of operation was 45 minutes and the mean length of chest tube duration 1.3 days. There were no deaths, no re-explorations or need to convert to an open thoracotomy. Staphylococcal pneumonia developed in one patient postoperatively requiring admission and intravenous antibiotics. One patient with systemic pulmonary hypertension was ventilator dependent for 48 hours. All patients, except two ventilator dependent patients, were intubated with a double lumen tube. CO2 insufflation at the rate of 2 L/min and pressure of 10 mmHg was used in all patients. Biopsy of at least two lobes was performed in all patients with resection of grossly abnormal lung. A single chest tube was left at the end of the procedure. The tissue diagnosis was interstitial fibrosis in 19 patients. Bronchiolitis obliterans with organizing pneumonitis (BOOP) was seen in 7 patients. Foreign body granulomas were seen in 8 patients. Allergic alveolitis was diagnosed in 4 patients. Emphysematous changes with pneumonitis was observed in 3, nonspecific pneumonitis in 2. Anthracosis, connective tissue disorder, leukemic infiltrate with interstitial fibrosis and CMV pneumonitis were observed in one patient each. The clinical diagnosis correlated with pathological diagnosis and intraoperative findings. Thoracoscopy is a safe and effective method for diagnosis of interstitial lung disease.     

Idiopathic nonspecific interstitial pneumonia/fibrosis: comparison with idiopathic pulmonary fibrosis and BOOP

Based on past difficulties in clinically differentiating patients with idiopathic pulmonary fibrosis (IPF), bronchiolitis obliterans-organizing pneumonia (BOOP), and nonspecific interstitial pneumonia/fibrosis (NSIP), which all manifest clinically as interstitial lung disease, experience with pathologically confirmed examples of the three diseases was reviewed to compare clinical profiles and prognosis and to define NSIP more clearly. Thirty-one patients (15 males and 16 females) were pathologically identified as NSIP and subclassified into either the cellular (n=16) or fibrotic group (n=15). All 31 patients were clinically considered to be idiopathic NSIP cases. Patients with idiopathic BOOP (n=16) and IPF (n=64) were compared with the NSIP patients. Subacute presentation of interstitial lung disease characterized both idiopathic NSIP and idiopathic BOOP. NSIP patients showed volume loss on a chest radiograph (29.0%) and honeycombing on a computed tomography scan (25.8%); these features were not found in BOOP patients. Bronchoalveolar lavage lymphocytosis was characteristic of both BOOP and NSIP. Two subgroups of NSIP can be recognized histologically: patients in the fibrotic group had a less favourable outcome than those in the cellular group. BOOP and NSIP had a more favourable outcome than IPF. In conclusion, idiopathic nonspecific interstitial pneumonia can be differentiated from other types of idiopathic interstitial pneumonia, both pathologically and clinically.     

Echocardiographic diagnosis of single coronary artery in double-outlet right ventricle

A 90-year-old woman was admitted to our hospital in December 1993 because of dyspnea on exertion and malaise. She had been well until October 1993, when she first noticed Raynaud's phenomenon, skin tightening, digital ulceration and scarring of her hands. On physical examination, generalized edema was found, along with acrosclerosis with contracture, especially in the fingers, wrists, and elbows. Inspiratory crackles were noted. A roentgenogram of the chest and an echocardiogram revealed pulmonary fibrosis, pulmonary congestion, and massive pleural and pericardial effusions. The pleural effusion was a transudate. Progressive systemic sclerosis was diagnosed, and furosemide and isosorbide were given. The edema and pulmonary congestion resolved, but the pleural and pericardial effusions did not. Prednisolone was given, which reduced the pleural effusion but not the pericardial effusin. The pleura and the pericardium are not usually involved in progressive systemic sclerosis, and this disease rarely occurs in patients over 70 years old. To the best of our knowledge, this was one of the oldest patients with progressive systemic sclerosis. The combination of massive pleural and pericardial effusions, and the advanced age of onset make the present case unusual.     

Elevation of antibodies to cytomegalovirus and other herpes viruses in pulmonary fibrosis

The aim of this study was to determine whether latent viral infection is associated with idiopathic pulmonary fibrosis (IPF), an interstitial lung disease whose aetiology remains to be elucidated. Cytomegalovirus (CMV) immunoglobulin G (IgG) and complement fixation (CF), Epstein-Barr (EB) viral capsid antigen (VCA) IgG, herpes simplex virus (HSV) IgG, adenovirus CF, and parainfluenza 3 virus haemagglutinin inhibition (HI) titres were measured in the serum from patients with pulmonary diseases. The study included five subject groups: 35 normal controls (aged (mean +/- SD) 38 +/- 17 yrs); 43 IPF (63 +/- 10 yrs), seven collagen vascular disease-related interstitial pneumonitis (CVD-IP) (62 +/- 12 yrs); 22 sarcoidosis (36 +/- 14 yrs); and 17 emphysema (66 +/- 11 yrs). Levels of CMV IgG in IPF (87.6 +/- 51.7) and CVD-IP (101.2 +/- 69.9) were significantly elevated compared to those in the control (30.9 +/- 24.1), sarcoidosis (34.4 +/- 38.3) and emphysema groups (40.3 +/- 24.6), whereas CMV immunoglobulin M (IgM) was generally below the limit of detection. Similarly, CMV CF titres in IPF and CVD-IP were elevated compared to those in other diseases. EB VCA IgG titres in IPF, CVD-IP and emphysema and HSV IgG in IPF were also elevated. In contrast, adenovirus CF and parainfluenza 3 HI titres demonstrated no significant difference among all of the groups investigated. Increases in cytomegalovirus immunoglobulin G and complement fixation titres with negative cytomegalovirus immunoglobulin M suggest that latent cytomegalovirus infection may be more prominent in idiopathic pulmonary fibrosis or collagen vascular disease-related interstitial pneumonitis. Together with the elevation of Epstein-Barr virus viral capsid antigen and herpes simplex virus immunoglobulin G in idiopathic pulmonary fibrosis and/or collagen vascular disease-related interstitial pneumonitis, it is rational to assume that these viruses may be implicated in the development of pulmonary fibrosis. Further study is necessary to investigate the relationship between latent viral infection and pulmonary fibrosis.     

Pulmonary edema associated with the Chinese medicine shosaikoto

A 45-year-old Japanese woman presented with a high fever, a nonproductive coughing, and severe dyspnea, and was admitted to another hospital. During the week prior to hospitalization, she had been given Shosaikoto for treatment of liver dysfunction of unknown etiology. Mycoplasma pneumonitis was initially suspected, so she was treated with antibiotics (clindamycin and minocycline) and received oxygen therapy. Pulmonary insufficiency worsened rapidly, and she was transferred to our hospital. On admission, a chest roentgenogram revealed bilateral alveolar infiltrates predominantly in the medial lung fields. Furosemide and high-dose methylprednisolone were immediately administered, but hypoxemia increased. When the PaO2 was 55.7 Torr while the patient breathed 100% oxygen, mechanical ventilation with positive end-expiratory pressure (PEEP) was started. Arterial blood-gas values improved dramatically, and the chest roentgenogram became clear. Our diagnosis of noncardiogenic pulmonary edema is based on the chest-roentgenographic findings, infiltration of inflammatory cells as seen in two lung-biopsy specimens and bronchoalveolar lavage fluid, the lack of findings of heart failure on physical examination and electrocardiography, and the good clinical response to PEEP. A positive lymphocyte stimulation test in response to Shosaikoto implicated this non-traditional herbal medicine as an etiologic factor in the non-cardiogenic pulmonary edema. Shosaikoto has been identified as the cause of interstitial pneumonia or eosinophilic pneumonia, but pulmonary edema associated with Shosaikoto has not been previously described. This case suggests that methylprednisolone treatment may be insufficient for Shosaikoto-induced pulmonary edema, and that mechanical ventilation with PEEP is very effective.     

A case of lymphocytic interstitial pneumonia

A 56-year-old female was admitted on November 1995 to our hospital because of the abnormal shadow on her chest X-ray. Although the chest X ray film revealed diffuse reticulonodular shadows in the bilateral lung fields and right hilar lymphadenopathy, she had not any complaints. Furthermore, mediastinal lymphadenopathy and polyclonal hypergammaglobulinemia were noted. For a further examination, transcutaneous thoracoscopic lung biopsy was performed on August 1996. The lung specimens showed a interstitial infiltration of small lymphocytes exclusively around bronchioles. And the diagnosis of lymphocytic interstitial pneumonia (LIP) was made. She had been suffered from bronchial asthma for 27 years. This is the first report of LIP accompanied with bronchial asthma. Its relationship between LIP and bronchial asthma remains unclear. In the 2 years of follow-up, she remained asymptomatic with unchanged chest radiogram. And her pulmonary function was preserved for the 2 years. But lymphocytic interstitial pneumonia may induce malignant lymphoproliferative disease potentially, we should carefully follow up.     

Correlation of cytologic and pathohistologic findings in ultrasonically-guided thin-needle biopsy of abdominal and retroperitoneal organs

A 71-year-old woman with loss of appetite was referred to our hospital. Imaging diagnosis revealed a large, cystically dilated left kidney with a solid tumor inside the cavity and right hydronephrosis. A chest X-ray revealed multiple metastatic lesions. A horseshoe kidney was found intraoperatively and left nephroureterectomy with partial cystectomy was performed. Histological diagnosis was poorly differentiated transitional cell carcinoma. She died of progressive pulmonary metastases 2 weeks after operation. This is the 19th case of a renal pelvic tumor associated with a horseshoe kidney reported in the Japanese literature. The diagnosis was confounded by the extreme dilation and deformity of the hydronephrotic kidney.     

Two cases of metastasizing benign leiomyomatosis from myoma uteri]

Case 1. A 50-year-old woman was referred to our hospital because of multiple bilateral small round lesion on chest radiography. She had undergone total hysterectomy for myoma uteri at the age of 33. She underwent thoracoscopic tumor excision at left lung. The lesion was proved benign. Right side lesions were laterly excised using thoracotomy. Case 2. A 49-year-old woman was referred to us because of two ovoid lesion at left lung area of chest radiography. She also had undergone total hysterectomy for myoma uteri at the age of 37. She underwent tumor extirpation using thoracotomy. All samples of two patients revealed, pathologically, lesions were consisted of benign spindle-like calls similar to those of myoma uteri. Therefore, we consider these lesions were pulmonary metastasis of myoma uteri. Myoma uteri has certain potential of metastasizing to the lung, in spite of benign disease.     

A rapidly progressive case of interstitial pneumonia

We treated a 51-year-old woman who had rapidly progressive respiratory distress with an interstitial shadow on chest roentgenogram. Pathologically, open lung biopsy specimens showed an acutely changed lesion such as interstitial inflammatory thickening, polypoid intraluminal organizing exudates, and also honeycombing which was not recognized on chest computed tomogram. These findings were considered unconformable to acute interstitial pneumonia (AIP), bronchiolitis obliterans organizing pneumonia (BOOP), and also usual interstitial pneumonia, although the clinical diagnosis was AIP or BOOP. We diagnosed a rapidly progressive interstitial pneumonia showing an acute lung injury pattern like AIP and BOOP. She showed significant recovery with corticosteroid and cyclophosphamide.     

A case of Sairei-to-induced pneumonitis diagnosed by lymphocyte stimulation test of bronchoalveolar lavage fluid

A 51-year-old woman was admitted with fever, cough and dyspnea. She had been taking Sairei-to, a traditional Chinese medicine, for 2 months. On admission, chest X-ray revealed a ground-glass appearance in the lung fields bilaterally and serum LDH was elevated. A differential cell count of the bronchoalveolar lavage fluid (BALF) showed that lymphocytes were increased, with a decreased ratio of CD4/CD8 cells. A lymphocyte stimulation test (LST) for Sairei-to using the lymphocytes in BALF was a positive, although a test of peripheral blood was negative. Sairei-to-induced pneumonitis was diagnosed based on the clinical course, laboratory findings, BALF cell analysis and LST of BALF. Only 4 cases of pneumonitis due to Sairei-to have been reported. This case suggests that LST of BALF is useful for diagnosing drug-induced pneumonitis.     

Hemodynamic effects of the somatostatin analog lanreotide in humans: placebo-controlled, cross-over dose-ranging Echo-Doppler study

Fas is expressed in various cells and transduces the cell death signal. p21 is a mediator of p53-dependent G1 arrest associated with deoxyribonucleic acid (DNA) damage. The upregulation of p53 and p21 associated with DNA damage in idiopathic pulmonary fibrosis has been described previously. In this study, p53, p21, and Fas expression and DNA damage were examined in interstitial pneumonia associated with collagen vascular diseases (CVD-IP). DNA damage was assessed by terminal deoxynucleotidyl transferase-mediated deoxyuridine triphosphate biotin nick end-labelling (TUNEL) and p53, p21 and Fas proteins were detected by immunohistochemistry in 13 cases of CVD-IP, 13 of sarcoidosis, seven of hypersensitivity pneumonitis (HP) and eight control patients with normal lung parenchyma. TUNEL-positive signals were found in bronchiolar or alveolar epithelial cells in 11 of 13 (85%) specimens of CVD-IP, but not in sarcoidosis, HP or controls, except for a case of chronic HP with pulmonary fibrosis. p53, p21 and Fas were detected in bronchiolar or alveolar epithelial cells in nine (69%), 10 (77%) and 12 (92%) of 13 specimens of CVD-IP, respectively, but not in sarcoidosis, HP or controls, except for a case of chronic HP. These results suggest that the upregulation of p53, p21 and Fas in bronchiolar and alveolar epithelial cells associated with deoxyribonucleic acid damage may participate in the process of pulmonary fibrosis in interstitial pneumonia associated with collagen vascular diseases and chronic hypersensitivity pneumonitis.     

Pigeon breeder's lung

In the acute form of pigeon breeder's lung, chest X-rays often reveal micronodular changes, but 7 of the 16 patients examined showed no changes. In the subacute form, chest X-ray changes are exceedingly rare: they occurred in only 1 of the 17 patients. Pigeon breeder's lung differs from the condition known as bird breeder's lung in that it turns much less often into a chronic condition (pulmonary fibrosis and emphysema).     

A case of pleural involvement followed by idiopathic retroperitoneal fibrosis

We report a rare case of pleural involvement followed by idiopathic retroperitoneal fibrosis in a 55-year-old man. He complained of right chest pain and his chest radiogram showed pleural thickening and calcification. Chest X-ray computed tomogram (CT) also showed a mass-like pleural thickening and a soft tissue density lesion surrounding the thoracic aorta, suggesting fibrosis. By open pleural biopsy, the mass-like pleural lesion was diagnosed as chronic pleuritis. Three years later, the patient experienced difficulty in urination, and bilateral hydronephrosis which was probably due to a retroperitoneal lesion was detected. Abdominal X-ray CT revealed a retroperitoneal lesion of soft tissue density comprised of a smooth and clearly delineated mass which encased the large vessels and ureters, compatible with retroperitoneal fibrosis. Although it was not possible to biopsy the retroperitoneal lesion, the lesion was clinically and radiologically thought to be retroperitoneal fibrosis. We could not determine the etiology of these findings, but such a case of pleural involvement followed by idiopathic retroperitoneal fibrosis was considered to be very rare.     

Response to treatment with progesterone in a patient with pulmonary lymphangioleiomyomatosis

Lymphangioleiomyomatosis (LAM) is defined as an abnormal proliferation of smooth muscles around lymphatics, venules, and brochioles. This article describes our experiences treating a 21-year-old, white female who experienced recurrent shortness of breath during air travel last year. Her episode was severe and the patient was transported to the hospital as soon as the airplane landed. Physical exam in the emergency room was significant for absent breath sounds on the right side and the chest X-ray revealed a pneumothorax. She required two chest tubes for complete lung re-expansion. Further evaluation showed an obstructive pattern and air trapping on pulmonary function tests. This patient was treated with Medroxyprogesterone acetate (MPA) for six months and subsequent pulmonary function tests revealed improvement in her condition.     

Pulmonary toxicity of drugs used to treat systemic autoimmune diseases

A number of drugs used to treat systemic autoimmune diseases can cause respiratory complications. These include bronchospasm, noncardiac pulmonary edema, interstitial pneumonitis and fibrosis, hypersensitivity, and numerous other disorders. Additionally, some of these drugs increase the risk of infections, particularly with opportunistic organisms. This article reviews the clinical presentation and mechanism of toxicity of drug related pulmonary complications.     

Allergic alveolar diseases. Problems in diagnosis and management

One of the diagnostic possibilities to consider when a patient presents with cough, fever, dyspnea, or pulmonary infiltrates is hypersensitivity pneumonitis. Some of the problems encountered in diagnosis of diffuse lung disease are illustrated in two case reports. In one of the cases, interstitial pneumonitis of insidious onset was attributed to inhalation of thermophilic organisms in moldy silage. In the other, the outstanding pathologic feature was bronchiolitis obliterans, and circumstantial evidence pointed to a home humidifier as the source of the problem.     

Extraperitoneal anterior approach to renal vessels in radical surgery of renal cancer

A 63-year-old female developed the chest wall tumor protruding into the right thoracic cavity. She consulted our hospital complaining of chest pain. 20 years before this episode, she had undergone tumor extirpation combined with the partial resection of adherent anterior chest wall and the diaphragm for large benign solitary fibrous tumor of the pleura. Because of the difficulty in making diagnosis by needle biopsy, operation (re-tumor extirpation combined with the chest wall) was done. Histological examination revealed that the tumor was malignant solitary fibrous tumor. We concluded that the tumor was the malignant recurrence of the benign solitary fibrous tumor resected 20 years before. Occasionally, benign solitary fibrous tumor of the pleura recurs and behaves more aggressively than primary, but such a long latent period as 20 years is quite uncommon. So we present the case and the review of the literature.     

Extrinsic allergic alveolitis: a review for the practitioner

Extrinsic allergic alveolitis (EAA) or hypersensitivity pneumonitis (HP) is a clinical syndrome characterised by an inflammatory, partly granulomatous, immune disorder involving interstitial and alveolar spaces secondary to inhalation of organic substances. The disorder is mainly due to occupational exposure, farmer's lung being the best-known example. Acute, subacute or chronic forms can be clinically differentiated. Given the fact that chronic forms may present a pattern of irreversible pulmonary fibrosis, clinicians must be aware of the diagnosis of EAA in every situation where the history shows a potential antigenic exposure. Prevention should be reinforced by increasing individual protective measures and by improving techniques used at the workplace.