Rituximab therapy in a patient with steroid-refractory bird fancier's lung.

A 62-year-old woman presented with a 3-month history of shortness of breath on exertion and dry cough. On examination, she was noted to have fine end-inspiratory crepitations over the upper zone of the lungs. Pulmonary function tests (PFTs) showed a restrictive defect. Initial chest radiography revealed diffuse reticular interstitial shadowing while high-resolution CT scan of the thorax showed fibrotic changes. Avian precipitins were also highly positive for pigeons, parrots and budgerigars. Taking into account these results, the patient was diagnosed with hypersensitivity pneumonitis. Antigen avoidance, oral glucocorticoids and azathioprine achieved an initial improvement in PFTs and symptoms; however, the patient still deteriorated, requiring long-term oxygen therapy. While working the patient up for lung transplantation, rituximab was given to good effect (acting as a bridging therapy) as it achieved symptomatic relief and stabilisation of her PFTs.

[Bird fancier's disease in a child: about a rare and unusual case]. / La maladie du poumon des éleveurs d´oiseaux chez un enfant: à propos d´un cas rare et inhabituel.

Bird fancier's disease falls within the spectrum of hypersensitivity pneumonitis secondary to immuno-allergic reaction to avian antigens. This occurs only rarely in children. It is found in two-thirds of patients with hypersensitivity pneumonitis. Diagnosis is not so easy. It is based on a body of clinical evidence. We here report the peculiar case of a 7-year-old girl with a family history of atopic disease initially treated as asthma based on the presence of wheezing and dyspnea and cough without improvement. The patient had worsening of symptoms such as dyspnea at rest complicated by cyanosis in respiratory distress. All of this took place in a context of alteration of general state. Clinical examination showed growth retardation, perioral cyanosis with digital hippocratism. Lung auscultation revealed bilateral crackling sounds. Chest X-ray objectified bilateral interstitial syndrome. Chest computed tomography (CT) scan showed diffuse ground-glass opacities. Laboratory tests revealed hypereosinophilia with hyper-IgE and excluded tuberculosis, cystic fibrosis, immune deficiency. In a second stage interview contact with birds was reported. Serological tests for bird fancier's disease were positive. The patient received inhaled corticosteroids associated with avoidance of exposure to birds. After a follow-up of 2 months, outcome was favorable. Given that the signs of bird fancier's disease are non-specific, this should be suspected in patients with respiratory symptoms associated with exposure to avian antigens.

Exacerbation of Previously Undiagnosed Bird Fancier's Lung by Pembrolizumab Therapy.

Immune checkpoint inhibitors have revolutionized cancer therapy. As the use of checkpoint inhibitors becomes widespread, the early recognition and treatment of their unique spectrum of adverse effects, called immune-related adverse events, become critical. Perhaps the most significant of these is the pulmonary toxicity currently described as "pneumonitis." However, little is known about the effects of immune checkpoint inhibitors on preexisting interstitial lung disease. We present a case of subclinical hypersensitivity pneumonitis that was exacerbated by pembrolizumab, a programmed cell death-1 inhibitor. This case illustrates a new immune-related adverse event and suggests that exacerbation of preexisting interstitial lung disease is a potential pulmonary toxicity from immune checkpoint inhibitor therapy.

Bird fanciers lung in mushroom workers.

Hypersensitivity pneumonitis has been described in mushrooms workers caused by exposure to mushroom or fungal spores in the compost used to grow mushrooms. We describe two mushroom workers who developed hypersensitivity pneumonitis due to exposure to avian proteins found in poultry manure which was used in producing mushroom compost. Both workers were employed in the compost production area. Both presented with typical features of HP. Both workers had negative serological and precipitin studies to Apergillus fumigatus, Saccarhopolyspora rectivirgula and thermophilic actinomycetes but had positive responses to poultry antibodies. Neither was exposed to mushroom spores. Both workers required initial therapy with corticosteroids. Relocation with avoidance of further exposure resulted in complete cure in one worker and change in work practice with the use of personal protections equipment resulted in the second workerclinical stabilisation. These are the first reported cases of bird fanciers lung in mushroom workers.

[Coronary artery bypass grafting in a patient with chronic bird fancier's lung].

Acute exacerbation is a devastating and fatal event of hypersensitivity pneumonitis. We report an 80-year-old man with chronic bird fancier's lung which acutely exacerbated after off-pump coronary artery bypass grafting. Our perioperative management included avoidance of inhalation of high concentration oxygen, administration of neutrophil elastase inhibitor, and long-term administration of antibiotics. Nevertheless, the patient experienced severe respiratory failure after influenza infection. High-resolution computed tomography (CT) showed new ground-glass opacities overlying fibrosis and traction bronchiectasis. We diagnosed acute exacerbation of hypersensitivity pneumonitis. Immediate steroid pulse therapy with empirical antibiotic administration was effective, and he recovered from critical respiratory failure. High-resolution CT was very helpful in the early detection of acute exacerbation of pneumonitis.

Environmental triggers of autoimmunity in anti-synthetase syndrome: the lungs under the spot light.

The prevalence of interstitial lung involvement in anti-synthetase syndrome (anti-SS) may be as high as 70% and is a major contributor to morbidity and mortality. Histidyl-tRNA synthetase (Jo-1) is the most common autoantigenic target among the aminoacyl-tRNA synthetases. We report two well documented anti-SS cases where it was observed significant exposure to a known inhaled offending antigen, development of a lymphocytic interstitial lung disease (ILD) and negative auto-antibodies, interpreted at first as hypersensitivity pneumonitis. Only after 14 and 30 months, respectively, the development of systemic symptoms compatible with anti-SS and anti-Jo-1 was observed. A growing body of evidence suggests that the lungs are the environment in which Jo-1 autoimmunity may be initiated and propagated. The description of the clinical and laboratorial evolution of these patients together with accumulated evidence of biological plausibility support the hypothesis that anti-SS can follow an episode of lung inflammation secondary to inhaled antigen exposure.

[A small bird gives severe consequences]. / En lille fugl med alvorlige konsekvenser.

In this case we report how a 31 year-old woman developed hypersensitivity pneumonitis due to her budgerigar. The suspicion that her pet was the reason for her symptoms was confirmed by the finding of specific IgG antibodies against budgerigar proteins. Clinical studies demonstrated a restrictive lung disorder, increased lymphocytes in bronchoalveolar lavage and ground glass changes in high-resolution computed tomography. The patient's symptoms receded over the course of a few days after removal of the animal, and subsequent IgG levels fell.

Not all children with nodular interstitial lung patterns in South Africa have TB-A rare case of paediatric "Bird Fanciers' disease".

Bird Fancier's disease is an allergic alveolitis that is rare in children. We describe the relevance of adequate history for making the diagnosis in children and the difficulty distinguishing this entity on chest radiographs and CT from imaging patterns caused by infections such as tuberculosis and HIV in developing countries. Pediatr. Pulmonol. 2011; 46:1134-1136. © 2011 Wiley Periodicals, Inc.

Pigeon breeder's lung in childhood: is family screening necessary?

In this paper, we report on clinical and laboratory findings of pigeon breeder's lung (PBL) in a mother and her 2 children: 2 with the chronic recurrent form, and 1 with the chronic insidious form without any symptoms or signs, who was diagnosed during family screening. The index case was diagnosed as having primary pulmonary hypertension on admission. However, on evaluation, the diagnosis was of pigeon breeder's lung in the index case and her symptomatic mother. High-resolution computerized tomography (HRCT) revealed interstitial lung disease in an asymptomatic sister of the index case. All cases responded to steroid treatment, and at the end of the third month, HRCT findings disappeared. In conclusion, in patients with acute respiratory symptoms and rapidly recovering pulmonary hypertension after hospitalization, PBL should be remembered. All family members of the index case should be screened by HRCT in terms of interstitial lung disease, even if they are asymptomatic.

Bird breeder's disease: a rare diagnosis in young children.

UNLABELLED: Bird breeder's lung disease is the most common form of hypersensitivity pneumonitis and is a rare entity in young children. We report three cases of children under 7 years of age in whom this diagnosis was confirmed early in the course of the disease. Three children aged 4.4 to 6.5 years presented with dry cough lasting for more than 1 month, dyspnoea, variable loss of appetite, weight loss, fatigue, fever and mild signs of respiratory distress. Chest X-ray films and CT scans showed a bilateral micronodular infiltrate. All three patients had strongly suggestive bronchoalveolar lavage fluid findings with lymphocytosis; two had elevated cell counts and decreased CD4/CD8 ratios. Lung biopsy confirmed the diagnosis in all children. Contact with allergens was identified in all children: two had spent holidays close to a farm in the previous month and one was living next to a pigeon house. In all children, avian precipitins were positive. The symptoms rapidly resolved after allergen avoidance and treatment with oral prednisone. Corticoid treatment was given between 11 and 15 weeks. One child relapsed and required long-term low-dose corticotherapy for 1 year. Lung function tests were normal in all three patients, 3.9 to 5.7 years after diagnosis. CONCLUSION: Bird breeder's lung disease is a rare entity but should be considered in young children presenting long lasting cough. While rapid allergen exclusion and start of treatment can avoid the evolution into irreversible lung fibrosis, clinical and biological evolution should be monitored carefully even after stopping corticoid treatment because of the possibility of relapse.

Rosella parrot exposure as a cause of bird fancier's lung.

A case of a 30-year-old man who presented with a 2-month history of progressively worsening dyspnoea, cough, and reduced exercise tolerance is discussed. A chest x-ray and computerized tomography of the chest suggested interstitial lung disease, which was confirmed on histology of an open lung biopsy. Careful questioning revealed that the patient had sustained close exposure to a rosella parrot acquired as a pet 9 months prior to presentation, which led to the diagnosis of bird fancier's lung. The case, investigations, and outcome are presented. This is followed by a discussion on extrinsic allergic alveolitis with particular emphasis on the importance of a complete social and environmental history in patients presenting with similar respiratory symptoms.

Hypersensitivity pneumonitis in children: pigeon breeder's disease.

Hypersensitivity pneumonitis (HP), an extrinsic allergic alveolitis, is a group of immunologically mediated, diffuse inflammatory lung parenchymal diseases. Pigeon breeder's disease (PBD) is one of the most common clinical forms of HP. It is caused by inhalation of various pigeon-derived materials and can present in different clinical forms. The diagnosis is difficult and the best diagnostic tool is correlation of onset of symptoms with time of exposure. Precipitating antibodies against the avian proteins form the characteristic precipitin reactions. The most effective treatment is avoidance of the antigens. Steroids, either systemic or topical (inhalational), can be used to treat HP. We report five children with different clinical forms of PBD in whom the diagnosis was confirmed by positive serum precipitating antibodies to avian proteins. Although the disease is rarely seen in children, it should be considered in any child with recurrent or unexplained respiratory symptoms. Nebulising steroids might be a useful alternative treatment for allergic alveolitis.

Clinical features of recurrent and insidious chronic bird fancier's lung.

BACKGROUND: Chronic bird fancier's lung (BFL) can be subgrouped into two types. One subgroup of patients develops interstitial pulmonary fibrosis after recurrent acute episodes (recurrent BFL), and the other subgroup of patients has no history of acute episodes but has slowly progressive chronic respiratory disease (insidious BFL). OBJECTIVE: To define the clinical characteristics of both types of BFL and to provide clues for diagnosis. METHODS: We performed a retrospective review of the medical records of patients with chronic BFL who were evaluated between October 1992 and March 2001 at the Tokyo Medical and Dental University Hospital in Japan. Patients were evaluated for their clinical characteristics, including history, laboratory, and immunologic findings; imaging; bronchoalveolar lavage; and histologic findings. RESULTS: Thirty-two patients with chronic BFL were included in this study; 15 patients had recurrent BFL and 17 had insidious BFL. The patients with recurrent BFL tended to breed dozens of pigeons in a loft, whereas the patients with insidious BFL were likely to be exposed to smaller birds kept indoors. Specific antibodies against pigeon dropping extracts or budgerigar dropping extracts were positive in 87% of the recurrent BFL cases and 35% of the insidious BFL cases. Antigen-induced lymphocyte proliferation was positive in more than 90% of both groups. The upper lung field was frequently involved in both groups as demonstrated by chest radiographic findings. In all of the patients with insidious BFL, the diagnosis was confirmed by positive laboratory-controlled inhalation test results. CONCLUSIONS: Insidious BFL may be misdiagnosed as idiopathic pulmonary fibrosis if a careful history is not taken and antigen-induced lymphocyte proliferation, careful imaging evaluation, and laboratory-controlled inhalation challenge testing are not conducted. In contrast, the clinical findings of recurrent BFL are consistent with hypersensitivity pneumonitis induced by other antigens.

[Nocardia otitidiscaviarum, cutaneous infection in a patient receiving long-term corticosteroid treatment]. / Nocardiose cutanée à Nocardia otitidiscaviarum chez un patient traité par corticothérapie au long cours.

A 62-year-old man, under long-term corticosteroid therapy for pigeon breeder's disease, was admitted to endocrinology disease department for cutaneous abscess on back, limbs and scalp. Culture of various bacteriological samples (cutaneous abscess, blood culture) isolated Nocardia otitidiscaviarum. The patient was treated by trimethoprime-sulfametoxazole during several weeks with abscess disappearance. Our laboratory quickly identificatied a bacteria belonging to the Nocardia genus, with simple technique, later confirmed by a specialized laboratory (Pr. Boiron Claude Bernard University Lyon I) with identification of Nocardia otitidiscaviarum. The proof of pulmonary nocardiosis could not be established despite the existente of several risk factors. Prognosis is poor for immunocompromised patients, but the secondary cutaneous dissemination phase presented a favourable evolution under antibiotic therapy.

[A case of bird fanciers' disease caused by parrot droppings].

A 50-year-old woman who had been treated for Parkinson's disease was admitted to our hospital because of exertional dyspnea and weight loss. Her chest computed tomography showed diffuse ground glass opacities in both lung fields. Bronchofiberscopy was performed, and the bronchoalveolar lavage fluid showed an elevated lymphocyte count. The serum Ouchterlony test yielded positive results for parrot droppings as well as for pigeon serum and droppings. She had had a parrot for several years, but no pigeon, and so we diagnosed bird fanciers' disease associated with parrot droppings. This responded well to steroid therapy. This case of bird fanciers' disease was caused by the droppings of only one parrot.

A family with extrinsic allergic alveolitis caused by wild city pigeons: A case report.

We describe a family in which the mother died of unresolved lung disease and whose 5 children, some of whom had previous signs of asthma, were subsequently affected by extrinsic allergic alveolitis caused by contact with wild city pigeon antigens. The children received systemic corticosteroids for 1 month and inhaled steroids for 24 months, while antigen exposure was reduced as much as feasible. This was followed by a quick clinical recovery and a slow normalization of chest radiographs and pulmonary function indices, especially of diffusion capacity, during a follow-up of 24 months. Because pigeon-breeder's lung caused by free-roaming city pigeons has not been previously described, it remains unclear whether this family developed the disease because of high antigen exposure or because of increased susceptibility. None of the supposedly high-risk human leukocyte antigen types were found in the children. Whether human leukocyte antigen B7 in 1 child played a role in the course of the illness remains speculative. It is unknown to what extent pigeon-breeder's lung caused by nondomestic birds remains undetected and misdiagnosed as difficult or steroid-resistant asthma. The question remains whether free-roaming city pigeons are indeed a public health risk. We suggest that atypical outdoor antigens be considered in all patients with nonresolving chest disease or therapy-resistant asthma.

[Extrinsic allergic alveolitis. Clinical experience at the Instituto National de Enfermedades Respiratorias (INER)]. / Alveolitis alérgica extrínseca. Experiencia clínica del INER.

Extrinsic allergic alveolitis is an interstitial lung disease caused by exposure to a variety of inhaled antigens. In Mexico, the most frequent form is due to the inhalation of avian antigens, markedly pigeon proteins. Depending on type and time exposure, the disease presents different clinical forms usually characterized by progressive dyspnea, ground glass or reticulonodular images on chest x rays, a restrictive functional pattern, rest hypoxemia worsening with exercise, and increase of T lymphocytes in bronchoalveolar lavage with an inversion in the helper/suppressor ratio. In this paper, we discuss a 15-year experience with this pathological problem in Mexico, emphasizing the differences with this disorder in Caucasian populations. Generally, our patients display a chronic form of the disease, which evolves to fibrosis in about one-half of the patients. In this sense, the diagnostic, prognostic, and therapeutic focusing exhibit different elements, and thus the development of clinical and basic research is strongly required.

[Asthmatic child without asthma]. / Een astmatisch kind dat geen astma heeft.

Two boys aged 13 and 7 years, displayed chronic coughing, dyspnoea on exertion, anorexia, weight loss, and fatigue. At first a diagnosis of asthma was made. However, a correct interpretation of anamnestic and clinical features, laboratory findings and radiographic results led to the diagnosis of 'pigeon breeder's disease' in both cases. Both patients recovered after drug treatment and avoidance of re-exposure to pigeon antigen.