Plastic Bronchitis: A Case Report

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 fi ve 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 diffi culty in diagnosing the disease and distinguishing associated pathologies; (iii) disseminate the disease to health professionals; (iv) better understand the available therapies; (v) evaluate the effi cacy of the disease treatment. Case Report Plastic Bronchitis: A Case Report 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 Dates: Received: 17 July, 2017; Accepted: 17 August, 2017; Published: 18 August, 2017 *Corresponding author: 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: Fernando AL Marson, Department of Medical Genetics, School of Medical Sciences, State University of Campinas, Tessália Vieira de Camargo, 126, Cidade Universitária Zeferino Vaz, Zip Code: 13083-887, Campinas, SP, Brazil, E-mail: https://www.peertechz.com


Introduction
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][2][3][4][5][6][7][8][9][10][11].

Clinical observation
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 identifi ed 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 [C 5 H 9 NO 3 S]. 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 ventilatorydependent 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  2 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 fi rst 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 fi brinopurulent serous exudate. After the second bronchoscopy, DOI: http://dx.doi.org/10.17352/2455-8141.000020 the elimination of abundant secretion and bronchial cast (approximately fi ve cm in length) were identifi ed. 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: 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 fi rst 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 fi gure 1.

Discussion
In the scientifi c literature, the prevalence of plastic bronchitis is unknown, arising mainly from the misdiagnosis in most patients [12,13]. In addition, the knowledge of the disease is based primarily on case reports [3,[6][7][8]12,[14][15][16][17][18][19][20][21][22][23] and the diagnosis of plastic bronchitis is often held during an autopsy after death by respiratory failure [13]. The disease affects all age groups, with a predominance of cases in women [12], a fact not yet clarifi ed in the literature.
The main characteristic of plastic bronchitis is the formation and expectoration of bronchial casts, which can vary in sizes, from small segments up to larger sizes capable of obstructing the airway of an entire lung. Bronchial casts consist of varying proportions of fi brin, mucin and cellular material. The sputum of bronchial casts varies in frequency and duration, and may occur hemoptysis [12].
In some cases of plastic bronchitis occurs spontaneous elimination of bronchial cast, which can complicate and delay the diagnosis. However, in our case report, the patient showed spontaneous elimination of bronchial cast that lasted for about a year.
The clinical pattern of the disease presents wide variability. However, chronic cough and dyspnea are usually observed, being wheezing the most common fi nding of physical examination. Radiological examination often identifi es the location of the bronchial impaction, with atelectasis or infi ltrators, predominantly in lower lobes [12]. Plastic bronchitis can present mild medical conditions with clinical recovery, as in our case report. However, in severe cases, airway obstruction and severe atelectasis can occur, leading to respiratory failure [12,13].
The High-Resolution Computed Tomography allow, in some cases, the visualization of bronchial casts in the airways of larger caliber. However, the diagnosis is usually confi rmed by bronchoscopy for demonstrating the airway obstruction by bronchial cast [12]. Interestingly, on the histological classifi cation, type 1 casts are associated with, primarily, the pathologies affecting the bronchial tubes (i.e., asthma and allergy); whereas type 2 casts are associated with congenital cyanotic heart disease and idiopathic plastic bronchitis cases. The ratings present therapeutic value or limited prognosis [13].
Diseases probably associated with plastic bronchitis are briefl y presented in table 1. However, cardiac changes and pulmonary interaction that result in plastic bronchitis are not well known, but they can be associated with abnormalities in the tissue factor [13]. Moreover, it should be considered that, recently, plastic bronchitis can be triggered by common respiratory infections and cause atelectasis, even in healthy children [6].
The assessment of treatment response in plastic bronchitis is restricted, considering that the disease is rare and the patients received different medications, being diffi cult to determine which therapy was effective [13].
The treatment of plastic bronchitis includes acute therapy to assist the removal and expectoration of bronchial casts and short or long-term treatments that address the hypersecretory process [12]. However, if plastic bronchitis can be a complication of an underlying disease, the underlying condition must be treated to eliminate the formation of bronchial casts.
Drugs used for treating plastic bronchitis present low scientifi c evidence. In short, for the different drugs already used, we have the following conclusions, mainly, obtained in the consensus of the European Respiratory Society (ERS) (2013) [13] and in the international registry data of plastic bronchitis (    agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work were appropriately investigated and resolved.