Cigarette smoking-induced acute eosinophilic pneumonia: a case report including a provocation test

The mechanism and cause of acute eosinophilic pneumonia are largely unknown. Many factors including the smoking of cigarettes have been suggested, but none have been proven to directly cause acute eosinophilic pneumonia. The authors report a case of acute eosinophilic pneumonia in a young Asian male who recently started smoking. The diagnosis was made based on his clinical course and results of chest radiography, lung spirometry, bronchoalveolar lavage, and transbronchial lung biopsies. After administration of methylprednisolone, his clinical course rapidly improved. A provocation test was designed to establish a connection between cigarette smoking and the development of acute eosinophilic pneumonia. After the provocation test, the patient showed identical symptoms, increase in sputum eosinophils, and worsening of pulmonary function. The results of the provocation test suggest that smoking may directly cause acute eosinophilic pneumonia, and support previous reports of cigarette smoking-induced acute eosinophilic pneumonia.     

Allergenicity of the osmophilic fungus Aspergillus restrictus evaluated by skin prick test and radioallergosorbent test

Recently large amounts of Aspergillus restrictus, a species of osmophilic fungi, have been detected in house dust using culture media with low water activity. But little attention has been paid to this fungus as an allergen. In the present study, the authors examined the allergenic activity of A. restrictus by skin prick tests and radioallergosorbent tests (RAST) on 94 asthmatic patients (mean age 12.0, range 3-18). Aspergillus fumigatus, Alternaria alternata and house dust were used for comparison. In the skin prick tests, A. restrictus, A. fumigatus, A. alternata and house dust elicited positive reactions in 8 (8.5%), 8 (8.5%), 15 (16.0%) and 69 (73.4%) patients, respectively. RAST showed positive reactions in 27 (28.7%) subjects for A. restrictus, 22 (23.4%) for A. fumigatus, 35 (37.2%) for A. alternata, and 75 (79.8%) for house dust. These results indicated that some asthmatic individuals showed immediate-type hypersensitivity to A. restrictus, and the prevalence of hypersensitivity of A. restrictus determined by skin prick tests and RAST was comparable with that of A. fumigatus but lower than that of A. alternata or house dust. This indicates that this fungal species may be of importance as a causative agent in atopic diseases.     

Chronic myelomonocytic leukemia in a patient with a familial t(6;16)(q13;q22) translocation

A 75-year-old man with chronic myelomonocytic leukemia was found to have a constitutional t(6;16)(q13;q22) translocation, as did his healthy daughter. Chromosomal in situ hybridization studies of the daughter's lymphocytes did not indicate translocation-mediated interruption of the metallothionein gene cluster, at 16q22, although this locus has been reported to be involved in the eosinophilic variant of acute myelomonocytic leukemia. Lymphocytes from the daughter and from the patient's brother (who had a normal karyotype), had no increased fragility at 16q22. The findings do not provide evidence for an association between the familial chromosome abnormality and this patient's leukemia.     

Evolution to eosinophilic leukemia with a t(5:11) translocation in a patient with idiopathic hypereosinophilic syndrome

The idiopathic hypereosinophilic syndrome (HES) comprises a diverse group of diseases that may ultimately lead to multiorgan dysfunction and death. We present a case of a man who was followed for over 9 years with HES that underwent malignant transformation to acute leukemia with eosinophilic features. The patient's clinical acceleration was accompanied by the development of a malignant clone that was identified with banding techniques as 46,XY,t(5:11)(p15;q13). Electron microscopy reaffirmed findings reported in earlier cases of eosinophilic leukemia. At no time during his illness were cytotoxic drugs administered. In addition to delineating the natural evolution and cytostructural details of the case, we emphasize the role of cytogenetics in the predicting of malignant variants of the hypereosinophilic syndrome and in identifying eosinophilic leukemia.     

Smoking-Induced Acute Eosinophilic Pneumonia in a 15-year-old Girl: A Case Report

Acute eosinophilic pneumonia is a very rare disease that is characterized by acute febrile respiratory failure, diffuse bilateral infiltrates on chest X-ray, and eosinophilia in bronchoalveolar lavage fluid in the absence of infection. We present the case of a 15-year-old girl diagnosed with smoking-induced acute eosinophilic pneumonia. A previously healthy young girl with a 1-day history of fever presented with cough, dyspnea, and diffuse bilateral infiltrates on chest X-ray. She had started smoking only 3 weeks before presentation. She was diagnosed by bronchoalveolar lavage fluid tests and lung biopsy and dramatically improved after steroid treatment. We emphasize that acute eosinophilic pneumonia must be considered when acute pneumonia does not respond to broad-spectrum antibiotics. Effective treatment and prompt institution of therapy can obviate unnecessary morbidity and mortality.     

A case of chronic eosinophilic pneumonia with significant elevation and variation of serum parainfluenza virus 3 antibody titer]

A 67-year old man visited our hospital because of cough and sputum. Chest radiograph revealed various consolidations of the bilateral lung fields. Eosinophilic pneumonia was diagnosed by transbronchial lung biopsy speciments. The clinical course and the treatment with corticosteroids improved chest radiographic findings quickly confirmed the diagnosis of chronic eosinophilic pneumonia. Based upon the significant elevation and variation of serum parainfluenza virus (PIV) 3 antibody titer, it was suggested that PIV 3 caused the chronic eosinophilic pneumonia.     

Acute eosinophilic pneumonia associated with amitriptyline in a hemodialysis patient.

Drugs are well known causes of eosinophilic lung disease. In many patients, drug-induced eosinophilic lung disease presents with transient eosinophilic infiltrates that disappear after discontinuation of the drug. Some patients, however, experience a fulminant, acute eosinophilia-like disease. Recently, we experienced a case of amitriptyline-associated acute eosinophilic pneumonia with respiratory failure in a diabetic hemodialysis patient. Eight days after treatment with amitriptyline, sudden fever, chill, dry cough and dyspnea developed. Subsequently, multiple patch consolidations appeared on the chest radiographs. Bronchoalveolar lavage (BAL), established a diagnosis of acute eosinophilic pneumonia. After immediate discontinuation of amitriptyline, a rapid clinical and radiological improvement was observed. The present case indicates that the possibility of acute eosinophilic pneumonia should be fully considered in dialysis patients developing unexplained respiratory symptoms while on amitriptyline therapy.     

Characterization of monoclonal antibodies to an antigenic protein from Stachybotrys chartarum and its measurement in house dust

Using sera from atopic patients we have isolated an extracellular protein, which is antigenic in humans, from Stachybotrys chartarum sesu lato. Here we report the production of monoclonal antibodies to the protein and the development of a sensitive and specific assay to the target protein as well as analyses in house dust samples spiked with spores. The detection limit for the target antigen in house dust was approximately 0.2 ng/g dry weight house dust. This detection limit is comparable to those for house dust mite allergen and the allergen of the fungus Aspergillus fumigatus but lower than that for the fungus Alternaria alternata.     

Chronic eosinophilic pneumonia with subpleural curvilinear shadow

We report a rare case of chronic eosinophilic pneumonia with subpleural curvilinear shadow. CT scan showed a patchy consolidation in the bilateral upper lungs. In addition, subpleural curvilinear shadow was found in the bilateral upper lungs. A bronchoalveolar lavage obtained from the right middle lobe showed 25 % eosinophils. Although very rare, we should therefore keep in mind that patients, who have patchy consolidation with areas of subpleural curvilinear shadow in the bilateral upper lungs, may have chronic eosinophilic pneumonia.     

Elevated soluble ADAM8 in bronchoalveolar lavage fluid in patients with eosinophilic pneumonia

BACKGROUND: ADAM (a disintegrin and metalloprotease) family members, characterized by a metalloprotease and a disintegrin domain, are membrane-anchored glycoproteins involved in proteolysis and cell adhesion. ADAM8 might have an important role in allergic inflammation. It can cleave a variety of substrates and is a sheddase for VCAM-1 and CD23, the low-affinity IgE receptors. METHODS: To evaluate the contribution of ADAM8 to the pathogenesis of eosinophilic pneumonia (EP), we measured the concentrations of soluble ADAM8 (sADAM8) and its substrates, soluble VCAM-1 (sVCAM-1) and soluble CD23 (sCD23), in bronchoalveolar lavage fluid from patients with smoking-induced acute eosinophilic pneumonia (AEP), chronic idiopathic eosinophilic pneumonia (CEP), and drug-induced eosinophilic pneumonia (drug-EP). RESULTS: The sADAM8 and sVCAM-1 concentrations were increased in AEP and CEP. The sCD23 concentration was elevated in AEP. In AEP, but not CEP, the sADAM8 concentration significantly correlated with those of both sVCAM and sCD23. CONCLUSION: The pathogenesis of AEP, CEP, and drug-EP was distinct with regard to ADAM8. Our results are the first to associate ADAM8 with eosinophilic responses and lung inflammation in humans.     

Acute eosinophilic pneumonia accompanied by mediastinal lymphadenopathy and thrombocytopenia

Acute eosinophilic pneumonia, which was described in 1989, is thought to represent a hypersensitivity reaction to unidentified inhaled antigens. Here, we present a case of a marble mine worker with acute eosinophilic pneumonia complicated with mediastinal lymphadenopathy, neutrophilia, thrombocytopenia and acute respiratory distress syndrome.     

Detection of IL-5 and IL-1 receptor antagonist in bronchoalveolar lavage fluid in acute eosinophilic pneumonia

BACKGROUND: Acute eosinophilic pneumonia is an idiopathic cause of respiratory failure, characterized by very high numbers of alveolar eosinophils without significant blood eosinophilia. OBJECTIVE: The purpose of this study was to determine which cytokines are associated with acute eosinophilic pneumonia. METHODS: Soluble IL-1 type II receptor and the cytokines IL-1β, IL-1ra, IL-3, IL-5, granulocyte-macrophage colony-stimulating factor, and tumor necrosis factor-α were measured in serum and in bronchoalveolar lavage fluid from two patients with acute eosinophilic pneumonia during both acute and convalescent phases. RESULTS: Compared with patients with adult respiratory distress syndrome, the patients with acute eosinophilic pneumonia had high bronchoalveolar lavage fluid levels of IL-5, IL-1ra, and soluble type II IL-1 receptor but not IL-1β, tumor necrosis factor-α>, IL-3, or granulocyte-macrophage colony-stimulating factor. Bronchoalveolar lavage fluid levels of IL-5 and IL-1ra fell after resolution of symptoms. In the serum of patients with acute eosinophilic pneumonia, IL-5 was not detectable, and IL-1ra was initially high but fell after corticosteroid treatment. CONCLUSION: Acute eosinophilic pneumonia is characterized by locally high levels of IL-5, IL-1ra, and soluble type II IL-1 receptor in the alveolar space. (J ALLERGY CLIN IMMUNOL 1996;97:1366-74.)     

Eosinophilic pneumonia-like areas in idiopathic usual interstitial pneumonia.

Usual interstitial pneumonia is the most common idiopathic chronic interstitial pneumonia, characterized by a temporally heterogenous pattern of interstitial injury with interstitial mononuclear infiltrates, septal fibromyxoid nodules, and parenchymal scarring. This report details the presence of focal eosinophilic pneumonia in six cases of usual interstitial pneumonia in the absence of known causes of this reaction. The relationship of eosinophilic infiltrates in usual interstitial pneumonia with regard to pathogenesis, differential diagnosis, and prognosis is discussed.     

Interrelationship of chronic eosinophilic pneumonia, bronchiolitis obliterans, and rheumatoid disease: a hypothesis.

Three patients with histologically proven bronchiolitis obliterans are presented, two of whom had rheumatoid disease. All three patients had, in addition, clinical and radiological evidence of chronic eosinophilic pneumonia; open lung biopsy in two showed coexistent features of chronic eosinophilic pneumonia and bronchiolitis obliterans. The association of both rheumatoid disease are chronic eosinophilic pneumonia with bronchiolitis obliterans in these patients may simply be coincidental, but the striking similarities between the cases suggest that a real interrelationship of these disease entities may exist.     

IgE antibodies against bovine serum albumin in a case of eosinophilic gastroenteritis

Eosinophilic gastroenteritis is a disease characterized histologically by an eosinophilic infiltration of the gut. The cause of this disease remains unclear, although both food allergy and food intolerance have been implicated in its pathogenesis. We report the case of a 22-year-old man in whom gastrointestinal symptoms first appeared in childhood, with involvement of mucosa and muscularis layers of stomach and bowel. He presented high IgE blood levels, and his prick test was positive to bovine, pig, and lamb sera. Immunoblots from calf, pig, and lamb sera, incubated with the patient's serum and revealed by autoradiography, demonstrated the presence of a 65-kDa protein band that was recognized by IgE antibodies but not by IgG. This band corresponded to bovine serum albumin, while IgE did not show reactivity with human albumin. These data suggest a possible role for IgE-mediated hypersensitivity mechanisms in the pathogenesis of eosinophilic gastroenteritis.     

Total serum IgE levels in eosinophilic lung diseases]

BACKGROUND: We previously reported elevated levels of total serum IgE in patients with asthma, regardless of their atopic status. We hypothesized that certain factors inherent to asthma may contribute to this non-specific elevation of total serum IgE. In the current study, to evaluate the role of eosinophils in the regulation of total serum IgE, we examined whether peripheral blood eosinophil count is associated with total serum IgE level in patients with eosinophilic lung diseases. METHODS: Ninety-nine healthy controls, 277 patients with asthma, 15 patients with acute eosinophilic pneumonia, 21 patients with chronic eosinophilic pneumonia were studied for total serum IgE levels and peripheral blood eosinophil counts. RESULTS: Patients with acute or chronic eosinophilic pneumonia had significantly increased total serum IgE levels compared with healthy controls regardless as atopic status (p<0.001). In non-atopic subjects with eosinophilic lung diseases, total serum IgE level was significantly correlated with peripheral blood eosinophil count (r=0.42, p<0.001, n=57). CONCLUSION: Our findings suggest that, in addition to antigen-specific IgE production, non-specific IgE production may contribute to elevated levels of total serum IgE in patients with asthma or eosinophilic pneumonia. An increased number of activated eosinophils may underlie an increased total serum IgE level in these conditions.     

Acute eosinophilic pneumonia as a reversible cause of noninfectious respiratory failure

Although chronic eosinophilic pneumonia is a well-known disorder, acute eosinophilic pneumonia has not been as well characterized. We describe the clinical features, results of bronchoalveolar lavage, and follow-up studies of four patients with acute eosinophilic pneumonia. The patients presented with an acute febrile illness, severe hypoxemia (partial pressure of arterial oxygen less than 60 mm Hg), diffuse pulmonary infiltrates, an increased number of eosinophils (mean +/- SEM, 42 +/- 4.8 percent) in bronchoalveolar-lavage fluid, and an absence of infection and previous atopic illness. The illness resolved rapidly after treatment with erythromycin and corticosteroids. The patients received doses of oral prednisone that were tapered over 10 days to 12 weeks, and none have relapsed since the steroids were discontinued. After a minimum follow-up period of five months, clinical evaluation, chest radiography, and pulmonary-function tests have shown no residual abnormalities attributable to the acute eosinophilic pneumonia. Follow-up bronchoalveolar lavage has demonstrated less than or equal to 1 percent eosinophils in all patients. We believe that we are describing an acute form of eosinophilic lung disease distinct from previously described syndromes. It can be diagnosed by bronchoalveolar lavage and seems to respond to treatment with corticosteroids.     

Allergy to iguana

BACKGROUND: Furry animals produce allergens that can cause allergic rhinitis and asthma. In contrast, scaly animals, such as lizards, are assumed not to be allergenic. OBJECTIVE: We sought to evaluate a 32-year-old man who complained of allergic rhinitis and asthma symptoms that occurred exclusively in his own home. He had dogs and cats at home but denied any increase in symptoms specifically associated with these pets. Skin prick testing initially performed to 42 common aeroallergens, including cat, dog, and house dust mite, elicited negative results. He later reported that the symptoms were worse on exposure to his pet iguanas. METHODS: Skin prick tests were subsequently performed to an extract made from scales from his pet iguana. Extracts were also prepared from several zoo reptiles. Immunoassays for IgE antibody, as well as IgE immunoblots, were performed by using these extracts and the patient's serum. RESULTS: The skin prick test result with the pet iguana scale extract was positive. The patient's serum contained IgE antibody to his own pet iguana and to a zoo iguana. CONCLUSION: Our patient's history, skin test results, and in vitro studies clearly demonstrate that he is allergic to iguana. Physicians should be aware that such allergy to scaly pets may occur and should not restrict history taking to questions about furry pets.     

De novo mutation causing X-linked hyper-IgM syndrome: a family study in Taiwan

X-linked hyper-IgM syndrome (XHIM) is a rare primary immunodeficiency disorder caused by mutations of the gene encoding the CD40 ligand (CD40L). It is characterized by recurrent infections with markedly decreased serum IgG, IgA and IgE levels but normal or elevated IgM levels. We report the clinical manifestations and complete immune studies in the first family with molecularly proven XHIM in Taiwan. A 5-month-old boy presented with rapidly progressive pneumonia which responded poorly to antibiotics. High levels of IgM and very low levels of IgG, IgA, and IgE were noted in his plasma specimen: IgM, 128 mg/dl; IgG, 18 mg/dl; IgA, 4 mg/dl); IgE, 1 IU/ml. Whole blood flow cytometry when he was 21 months old showed that only a small percentage (0.48%) of his in vitro-activated CD4+ T cells expressed CD40L. When he was 3 years old, repeated flow cytometry showed essentially the same result (0.4%), compared with his father's CD40L expression of over 85%. The patient's mother had moderately decreased CD40L expression (74.4%). Hyper-IgM syndrome was confirmed by CD40L mutation analysis in the boy, which revealed a Lys 96 stop (nucleotide A307T) in exon 2 of CD40L, with a truncated protein resulting in the loss of the entire TNF domain. His mother was a carrier and apparently the individual in whom the mutation originated. Eleven other family members, including the patient's father, sister, and grandmother, and the mother's sisters and their children, all had normal results on CD40L mutation analysis. The patient has remained without significant bacterial infection on a regimen of monthly IVIG infusion and oral trimethoprim-sulfamethoxazole for Pneumocystis carinii pneumonia (PCP) prophylaxis, although he has had recurrent oral ulcers and neutropenia. Bone marrow transplantation is planned.     

Successful Treatment of Chronic Eosinophilic Pneumonia with Anti-IgE Therapy

Anti-IgE therapy, using recombinant humanized anti-IgE antibodies, is clinically effective in patients with eosinophil-related disorders such as allergic asthma, allergic rhinitis, and chronic urticaria. Chronic eosinophilic pneumonia tends to respond promptly to systemic corticosteroid therapy, however; relapses are common following corticosteroid tapering. We treated two patients (17- and 19-yr-old males) of chronic eosinophilic pneumonia whose symptoms were cough and dyspnea on exertion. The symptoms were recurrent while tapering off corticosteroid. They were treated with anti-IgE antibody without recurrence for 2 yr and 15 months. Here, we first describe clinical experience of the 2 cases of chronic eosinophilic pneumonia.