Treatment of cast bronchitis with low‐dose oral azithromycin

Abstract

Cast or plastic bronchitis is an unusual disorder that is rarely encountered in the pediatric population. It is characterized by the expectoration of large, branching plugs of airway debris. These "casts" conform to the shape of portions of the tracheobronchial tree, and give the disorder its name. Cast bronchitis is typically seen in association with several primary pulmonary disorders and cyanotic congenital heart disease. It can be classified as inflammatory or acellular, based on the histologic characteristics of the casts. The presence of large, obstructive plugs filling the airways of lobes or entire lungs can result in a variety of clinical signs and symptoms, and may ultimately lead to respiratory failure and death. Conventional treatment of cast bronchitis has focused on the clearance of obstructing material from the airways combined with therapy for any underlying cardiopulmonary disease. Unfortunately, this approach has not proven very effective, and patient mortality remains high. We report on a case in which a patient with cast bronchitis was treated with long-term, low-dose oral azithromycin. This therapy resulted in clinical, spirometric, and radiographic improvement of the patient.

Spontaneous resolution of plastic bronchitis in a patient post hemi-Mustard/bidirectional Glenn atrial switch procedure in the double-switch operation for congenitally corrected transposition of great arteries after course of Augmentin

“…Oral macrolide antibiotics are described as treatment options [7]. However, Augmentin was used with our patient and a fast response ensued, which we think is worth reporting for possible future inclusion in medical therapy trials.…”

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

“…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.…”

“…Nearly month-long therapy of high doses ([1 mg/kg/day) resulted in fatal pulmonary hemorrhage, whereas low-dose therapy was well tolerated. Anti-inflammatory therapy use of corticosteroids and macrolides has been documented [2,57,58]. Macrolides have shown benefit in airway inflammation in other chronic lung disease [57].…”

“…However, specific benefit of clarithromycin in plastic bronchitis demonstrates conflicting results [36,57]. Low-dose azithromycin at a dose of 250 mg three times per week in conjunction with an oral corticosteroid 5-day course was shown to be effective in one case with eosinophilic and Charcot-Leydin cast [58]. Corticosteroid use also was successful for the treatment of an 8-year-old Fontan circulation patient with 1 % eosinophilia and casts showing mild eosinophilic infiltration [65].…”

Management of Plastic Bronchitis With Topical Tissue-type Plasminogen Activator

“…In general, patients with cyanotic heart disease have a poor prognosis, with mortality rates of 28% to 60%, whereas those with inflammatory disease have lower mortality (6%-50%). 3,13 Several therapies have been used for patients with cast bronchitis, including optimizing cardiac function, thoracic duct ligation, 14 bronchoscopic removal, 7,15-17 antiinflammatory medication, 18 and both mucolytics 12 and fibrinolytics, 4,19-21 but recurrences are common and mortality remains high.…”