A case of bronchial cast

Murata, Ryousuke; Ozaki, Hajime; Shiomi, Masashi; Fujita, Keisuke; Nakagawa, Keisuke; Kaji, Arito

doi:10.1007/bf01395716

Cited by 5 publications

(2 citation statements)

References 8 publications

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“…1,3,4,9,15,16 In pediatric patients with congenital heart disease, 15 cases of plastic bronchitis have been described previously in the literature (Table 1). Including our patient, 12 children with palliated single-ventricle physiology have been reported.…”

Section: Discussionmentioning

confidence: 99%

Treatment of Plastic Bronchitis in a Fontan Patient With Tissue Plasminogen Activator: A Case Report and Review of the Literature

et al. 2002

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ABSTRACT. Plastic bronchitis is a condition in which large, bronchial casts with rubber-like consistency develop in the tracheobronchial tree and cause airway obstruction. We describe a 4-year-old girl who had Fontan physiology and who developed plastic bronchitis and report for the first time the use of aerosolized tissue plasminogen activator for treatment of this condition. The literature is reviewed with emphasis placed on the occurrence of this disorder in patients with single ventricle physiology. Pediatrics 2002;109(4). URL: http:// www.pediatrics.org/cgi/content/full/109/4/e67; plastic bronchitis, bronchial cast, Fontan, congenital heart disease, tissue plasminogen activator.ABBREVIATION. t-PA, tissue plasminogen activator. P lastic bronchitis is a condition in which large, pale tan, bronchial casts with rubber-like consistency develop in the tracheobronchial tree and cause airway obstruction. 1 It is an unusual disorder that occurs in various disease states, and fewer than a dozen cases have been reported regarding patients with palliated congenital heart disease. We describe a 4-year-old girl who had Fontan physiology and who developed plastic bronchitis and report for the first time the use of aerosolized tissue plasminogen activator (t-PA) for treatment of this condition. The literature is reviewed with emphasis placed on the occurrence of this disorder in patients with single ventricle physiology. CASE REPORTOur patient's double-inlet left ventricle and L-transposition of the great arteries were diagnosed at birth. Her cardiac palliation performed at an outside institution consisted of a pulmonary artery band at 1 month of age; a bidirectional Glenn operation at 12 months; and a fenestrated, lateral tunnel Fontan operation at 26 months. Fenestration closure using a subcutaneous snare was performed 6 months later. Cardiac catheterization at 48 months of age revealed excellent hemodynamics without Fontan pathway obstruction. The mean pressure in the lateral tunnel was 10 mmHg, and there was no gradient to the left or right pulmonary arteries. The left ventricular end diastolic pressure was 4 mmHg, and the bulboventricular foramen was unobstructed.At 53 months of age, she presented to our institution with respiratory failure requiring mechanical ventilation. She improved with nebulized bronchodilator therapy and chest physiotherapy. This episode of respiratory decompensation was attributed to a mucus plug. During the next 4 months, she had 4 additional hospital admissions for perceived asthma exacerbations and pneumonia. Chest radiographs on each of these admissions revealed shifting infiltrates and atelectasis. A bronchoscopy performed during the third admission was unremarkable. The diagnosis of plastic bronchitis was made when the patient's parent brought an expectorated bronchial cast to the pediatrician after the fourth admission and bronchoscopy during a subsequent admission revealed a large bronchial cast. She was then treated on an outpatient basis with chest physiotherapy, inhaled steroids, al...

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Section: Discussionmentioning

confidence: 99%

Treatment of Plastic Bronchitis in a Fontan Patient With Tissue Plasminogen Activator: A Case Report and Review of the Literature

et al. 2002

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“…These casts may be associated with bronchopulmonary diseases of mucus hypersecretion, bronchopulmonary bacterial infections, congenital and acquired cardiac diseases or pulmonary lymphatic abnormalities [3]. Plastic bronchitis occurs in a wide variety of noncardiac conditions, including asthma [4][5][6][7], pneumonia [8], lymphangiomatosis [9][10][11], allergic bronchopulmonary aspergillosis [12], cystic fibrosis [13], bronchiectasis [7,14], acute chest syndrome associated with sickle cell disease [15], smoke inhalation [16] and idiopathic states [4,5,8,11,17,18]. The casts are of variable sizes and take the shape of the bronchi in which they formed.…”

Section: Discussionmentioning

confidence: 99%

A near lethal case of plastic bronchitis following truncal valve replacement

Lang

Champion

Cosper

et al. 2015

J Pediatr Intensive Care

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Plastic bronchitis is a condition in which large, rubber-like, bronchial casts develop in the tracheobronchial tree causing airway obstruction. It is an unusual disorder that occurs in various disease states. Most case reports of plastic bronchitis associated with congenital heart disease are in patients palliated with Fontan physiology. We describe a 13-year-old girl with DiGeorge syndrome and truncus arteriosus who underwent an uneventful truncal valve replacement. The child developed plastic bronchitis on postoperative day eight requiring extra-corporeal membrane oxygenation support and daily bronchoscopy to remove bronchial casts. Our patient did not have elevated systemic venous pressures or Fontan physiology. We speculate the etiology of plastic bronchitis may have been surgical trauma to the lymphatic channels surrounding the bronchi and disruption of pleural adhesions. A comprehensive evaluation revealed no specific cause and the diagnosis of idiopathic plastic bronchitis was made.

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