Andressa Oliveira Peixoto1,2*, José Dirceu Ribeiro1,2, Rafael Miranda da Costa3 and Fernando Augusto Lima Marson1,2,4*
1Department of Pediatrics, School of Medical Sciences, State University of Campinas, Cidade Universitária Zeferino Vaz, Barão Geraldo, Zip Code: 13083-887, Campinas, São Paulo, Brazil
2Centre for Research in Pediatrics (CIPED), School of Medical Sciences, State University of Campinas, Cidade Universitária Zeferino Vaz, Barão Geraldo, Zip Code: 13083-887, Campinas, São Paulo, Brazil
3Clinical Hospital of Sumaré, State University of Campinas, Av. da Amizade, 2,400, Jd. Bela Vista, Zip Code: 13175-49, Sumaré, São Paulo, Brazil
4Department of Medical Genetics, School of Medical Sciences, State University of Campinas, Cidade Universitária Zeferino Vaz, Barão Geraldo, Zip Code: 13083-887, Campinas, São Paulo, Brazil
Received: 17 July, 2017; Accepted: 17 August, 2017; Published: 18 August, 2017
Andressa O Peixoto, Department of Pediatrics, School of Medical Sciences, State University of Campinas, Tessália Vieira de Camargo, 126, Cidade Universitária Zeferino Vaz, Zip Code: 13083-887, Campinas, SP, Brazil, Tel: +55 19 35218983; Fax: +55 19 35218964; E-mail:
Peixoto AO, Ribeiro JD, da Costa RM, Marson FAL (2017) Plastic Bronchitis: A Case Report. Glob J Allergy 3(2): 022-026. DOI: 10.17352/2455-8141.000020
© 2017 Peixoto AO, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Plastic bronchitis is a rare disease characterized by the formation of bronchial casts that can cause partial or complete obstruction of the airway. In this case report, a patient aged four years, previously healthy, began a chronic cough and underwent repeated courses of antibiotic therapy, inhaled corticosteroids and imaging tests; until there was a spontaneous sputum of a bronchial tree-shaped cast with approximately five cm, making possible the diagnosis of plastic bronchitis, which in our case report has idiopathic etiology. Due to the rarity of the pathology, we believe in the importance of the case report that can help to: (i) better understand the clinical presentation of plastic bronchitis; (ii) understand the difficulty in diagnosing the disease and distinguishing associated pathologies; (iii) disseminate the disease to health professionals; (iv) better understand the available therapies; (v) evaluate the efficacy of the disease treatment.
Plastic bronchitis is a rare disease characterized by the formation of extensive bronchial casts, which can be gelatinous or rigid. Bronchial casts represent the exact format of the airway from which they are removed by sputum or bronchoscopy, and they tend to be large and more cohesive than mucous plugs [1-11].
Plastic bronchitis can be commonly associated with cyanotic congenital heart diseases, secondary acute chest syndrome to sickle cell disease and diffuse hypersecretory bronchial disorders, such as asthma, cystic fibrosis, bronchopulmonary aspergillosis, bacterial or viral respiratory infections, in addition to reports on cases of heart failure and pericardial effusion [1-11].
In our case report, a female patient, aged four years and six months, from the state of Minas Gerais, Brazil, was evaluated. The patient was healthy until the age of four years and three months, when she began a productive cough, accompanied by fever and wheezing, with no dyspnea. At that time, the presence of opacity in the left hemithorax was identified by the simple chest radiography and bronchopneumonia was diagnosed. The treatment of bronchopneumonia was held with the use of amoxicillin for 14 days, associated with N-acetylcysteine [C5H9NO3S]. After the treatment, there was partial improvement of the radiological image.
The patient remained symptom free during the two-week period after the treatment, when evolved, with ventilatory-dependent chest pain, with greater intensity in precordial region, associated with coughing and wheezing, but remaining afebrile. At that time, new chest X-rays showed left lobar atelectasis. Due to the clinical pattern, the patient was hospitalized to perform respiratory physical therapy, inhaled therapy with short-acting β2 agonist and antibiotic therapy for 12 days. Procedures were held at another hospital.
There was no radiological improvement with the interventions described, and bronchoscopy was indicated. The result of the first bronchoscopy showed no abnormalities. However, due to the present and constant radiological alteration, new bronchoscopy was indicated within 72 hours. In the second procedure, the presence of bronchial cast was evidenced, composed of amorphous, necrotic material and fibrinopurulent serous exudate. After the second bronchoscopy, the elimination of abundant secretion and bronchial cast (approximately five cm in length) were identified. Thereafter, the patient began to eliminate bronchial tree-shaped material after a cough crisis for a period of approximately one year.
Considering the possibility of plastic bronchitis diagnosis, the patient was referred to a Tertiary Hospital, for investigation, which included the analysis of underlying diseases.
In the Tertiary Hospital, a High-Resolution Computed Tomography of the chest was held, which showed:
(i) Total atelectasis of the left upper lobe, with volumetric reduction of the lung
(ii) Deviation of mediastinal structures to the left
(iii) Hilar prominence of difficult characterization to the left. For this reason, the radiology team indicated an exam with use of contrast.
The patient was submitted to rigid bronchoscopy in which bronchial casts or anatomical changes to lung segments on the left and right were not observed. However, it was identified high amount of whitish and thick secretion on the left. In the collection of bronchoalveolar lavage, the Staphylococcus aureus sensitive to oxacillin was identified in the routine diagnostic culture. In addition to the bacterial examination, analyses were performed for fungi and mycobacteria culture, and all the tests were negative.
After two months, a High-Resolution Computed Tomography of the chest with contrast was held. In the exam, atelectasis of the anterior segment of the left upper lobe and lingula was identified in the pulmonary parenchyma and was associated with bronchiectasis and parenchymal bands.
Since clinical symptoms persisted, the investigation of differential diagnosis or diseases known to underlie plastic bronchitis proceeded, which included the diagnostic analysis for:
I. Cystic fibrosis (determination of chloride ion concentration by the sweat test)
II. Allergic asthma [determination of immunoglobulin E (IgE), eosinophils and lung function performed by spirometry with bronchodilator therapy]
III. Tuberculosis (assessment by Mantoux test)
IV. Cardiopathy (assessed by echocardiography). However, there was normality in the examinations held for the diagnosis of underlying diseases.
In addition to the courses of antibiotic therapy that the patient received during the evolution of the disease, the use of long-acting inhaled corticosteroid and bronchodilator was indicated, due to the apparent possibility of asthma. A year after the procedures were held, the patient stopped presenting cough with mucus, and evolved without symptoms, although the etiology of plastic bronchitis has not been elucidated in the case.
Five years after the first scan, a new image exam was held, and it was noted:
(i) Pulmonary parenchyma with no changes
(ii) topography of the left lower lobe and lingula with discreet dilatation
(iii) Sparse bronchiectasis to the left. In addition, the evidence of pulmonary function remained within the normal range.
After the procedures, the patient continues to be accompanied at the Tertiary Hospital with no further complications and need for drug therapy.
The summary of the tests and its results are described in figure 1.
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