Rare case of Plastic bronchitis: A report

Published On 2019-10-17 12:30 GMT   |   Update On 2019-10-17 12:30 GMT

Dr Clara L. Grizales at Department of Paediatrics, Pediatric Pulmonology Service, Fundación Valle del Lili University Hospital, Cali, Colombia and colleagues have reported a rare case of Plastic bronchitis. The case has appeared in the Journal of Respiratory Medicine Case Reports.


Plastic bronchitis is a rare and underdiagnosed disease characterized by the formation and expectoration of bronchial casts of amorphous material, which can be potentially fatal. It is more frequent in the pediatric population. Symptoms can range from chronic cough and dyspnea to respiratory failure depending on the area of the compromised airway.




Patients of 4 years and 5 months old, with a history of complex congenital heart disease suspected from the prenatal period by fetal echocardiography that showed pulmonary atresia without ventricular septal defect treated with Glenn surgery at 8 months old and subsequently fenestrated Fontan procedure at 3 years of age. Chronically managed with carvedilol 1.12 mg every 8 hours, sildenafil 6 mg every 8 hours, bosentan 20 mg every 12 hours, enalapril 2.5 mg every 12 hours, furosemide 8 mg every 12 hours, acetylsalicylic acid 70 mg daily, warfarin with adjustment according to INR and spironolactone 20 mg every day. Without previous respiratory symptoms, consulted for 5 days of dry cough and respiratory distress, followed by expectoration of bronchial casts of whitish amorphous material accompanied by spots of blood. On physical examination, heart auscultation showed no gallop or murmurs, rhonchi were present in both lung fields, abdomen without masses and the extremities showed no abnormalities. Chest X-rays revealed reticular opacities predominantly bilateral at the bases. Due to the characteristics of the expectorated material, a diagnosis of PB was considered and the patient was hospitalized for management.


Laboratory exams at admission showed leukocytes 7160, neutrophils 59.6%, lymphocytes 30.7%, Hb14.4, Hct 43.4%, platelets 109.000, C-reactive protein 1.11, PT 30.3 seconds, PTT 40.4 seconds, INR 2.11, Alpha 1 Antitrypsin 1.5 (0.9–2), BUN 18, creatinine 0.4, sodium 136, potassium 3.52, chlorine 99.4, calcium 8.64, arterial gases with pH 7.406, PO 236.9, PCO 245.7, HCO 328.1 and negative blood cultures at 48 hours. Chest CT scan showed a post-surgical condition with permeable Fontan circulation, moderate stenosis of the preanastomotic right pulmonary artery, a decrease in the caliber of the distal branches without evidence of acute pulmonary thromboembolism with a pattern compatible with chronic thromboembolism, mosaic attenuation, bronchial occupation by long mucus plugs with secondary atelectasis, thin aortopulmonary collateral bronchial arteries (<1.5mm), tricuspid atresia, hypoplastic right ventricle and IAC without VSD. Studies for hypercoagulability evaluation in addition to normal coagulation factors VIII, V, VII, X, XI, and II were normal (see Fig. 1).




Treatment was initiated with oxygen therapy, micro-nebulizations with bronchodilator salbutamol prior to the nebulization of 3% hypertonic saline solution every 12 hours, in addition to nebulizations with dornase alpha 2.5 mg daily and nebulized heparin 5000 units every 8 hours. The patient persisted with cough, mobilization of pulmonary secretions, moderate dyspnea and expectoration of casts of gelatinous material for six days. Bronchoscopy was performed, the mucosa was found with edema and moderate erythema with remains of the thick mucous material on the carina and main bronchi as well as casts that were removed leaving the bronchial tree clean. The culture of the tracheal secretion reported a mixed flora. The macroscopic analysis of the expectorated sample evidenced a mucoid material that shaped the bronchial architecture. Microscopically, with hematoxylin and eosin staining and Movat staining, acellular mucoid material was observed which confirms bronchial cast type II (Fig. 2).



During hospitalization, cardiac catheterization was performed, showing a patent fenestrated Fontan, with low pulmonary vascular resistance and pulmonary pressure with Fio2 100%, and stenosis of the pulmonary artery for which stent placement was performed without complications. It was considered that the obstruction at the level of the pulmonary valve could be the trigger of PB. The patient continued with combined treatment and respiratory therapy achieving a saturation improvement and progressive reduction of the expectoration of casts, with discharge for outpatient management where, according to the evolution, the relevance of lymphatic embolization for long-term management of PB will be defined.


For more details click on the link: https://doi.org/10.1016/j.rmcr.2019.100876

Article Source : Respiratory Medicine Case Reports

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