Plastic bronchitis in children: A case series and review of the medical literature

Abstract

Plastic bronchitis is characterized by marked obstruction of the large airways by bronchial casts. We reviewed our experience and the literature to determine whether mortality rates are determined by underlying disease or cast type. We present 3 children with obstructive bronchial casts. One 3-year-old patient with Noonan's syndrome developed respiratory failure following surgery for tetralogy of Fallot requiring support with extracorporeal membrane oxygenation (ECMO) the first such case. There were 42 cases in the literature of children with plastic bronchitis. Casts may be divided into two types. Type I casts are inflammatory, consisting mainly of fibrin with cellular infiltrates, and occur in inflammatory diseases of the lung. Type II, or acellular casts, consist mainly of mucin with a few cells, and usually occur following surgery for congenital cardiac defects. Patients categorized by underlying disease included 31% with asthma or allergic disease, 40% with underlying cardiac defects, and 29% with other diseases. Mortality was 16%, but increased to 29% in patients with cardiac defects. Deaths occurred as long as 1 year after surgical repair for underlying defects. There were no deaths in patients with asthma. Life-threatening events were statistically higher in patients with cardiac defects (41%) than in those with asthma (0%, P = 0.02). Higher mortality in patients with type II casts compared to type I casts did not reach statistical significance (28% vs. 6%; P = 0.06). In conclusion, patients presenting with plastic bronchitis are at high risk for serious complications, especially with underlying cardiac disease.

Successful Management of Plastic Bronchitis in a Child Post Fontan: Case Report and Literature Review

“…Brogan et al reclassified casts depending on the disease states of asthma, cardiac, and idiopathic. Madsen et al expanded this classification to cardiac, lymphatic, asthmatic, and sickle cell-related disease states [1,2]. Madsen et al [2] formulated this classification based on details of previous case studies.…”

“…Madsen et al expanded this classification to cardiac, lymphatic, asthmatic, and sickle cell-related disease states [1,2]. Madsen et al [2] formulated this classification based on details of previous case studies. Cardiac patients described by them include eight Fontan patients, three Blalock-Taussig shunt patients, and one Glenn shunt patient.…”

“…Three additional cases of pericardial effusions and plastic bronchitis as a complication of Fontan operation have been detailed [2,13,33]. Bronchial casts in Fontan patients also have been eliminated by methods of fenestration [32][33][34][35], stent placement [36], atrial pacing [2,37], anticoagulation therapy [38], and pulmonary vasodilation [39][40][41].…”

“…Case studies have described the use of inhaled therapies [2,12,[53][54][55][56], anti-inflammatories [57,58], diuretics [38], and surgical palliation [34,35,59,60]. Brogan et al [2] incubated a cast from a Fontan patient in vitro with acetylcysteine, DNase, urokinase, and tPA, demonstrating that acetylcysteine had the greatest effect in dissolving the casts.…”

“…The mucoid impaction with cast formation occludes the major bronchi with firm, gelatinous material [1]. Seen in asthma and sickle cell disease, the morbidity and mortality is greater in congenital heart disease [2]. Clinical management for both acute and chronic plastic bronchitis remains difficult and poorly defined.…”