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Ann Thorac Surg 2007;83:1884-1886
© 2007 The Society of Thoracic Surgeons
a Department of Pathology, Frontier Lifeline, and Dr KM Cherian Heart Foundation, Chennai, India
b Department of Cardiology, Frontier Lifeline, and Dr KM Cherian Heart Foundation, Chennai, India
c Department of Cardiothoracic Surgery, Frontier Lifeline, and Dr KM Cherian Heart Foundation, Chennai, India
Accepted for publication December 13, 2006.
* Address correspondence to Dr Nayar, Frontier Lifeline & Dr K M Cherian Heart Foundation, R 30 C Ambattur Industrial Estate Rd, Mogappair, Chennai 600 101, India (Email: divya_s35{at}hotmail.com).
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Seear and colleagues [2] proposed a classification of bronchial casts as type I (cellular) or type II (acellular). Type I casts occur in the setting of underlying pulmonary inflammatory disease and consist of fibrin with inflammatory cells. Type II casts are seen more commonly in patients with a history of palliative surgery for cyanotic heart disease and consist of mucin without an inflammatory infiltrate. This case report describes a type I cast in a patient with completion Fontan procedure and the treatment modalities with reference to the pathogenesis of the disease.
An 8
-year-old boy was diagnosed 8 years earlier with a double-outlet right ventricle, large inlet ventricular septal defect, and severe pulmonary stenosis. A bilateral Glenn anastomosis was done at that time. This was followed by an extracardiac Fontan procedure with fenestration in the conduit 17 months previously. Postoperative bilateral chylothorax developed; more on the right side. The chylothorax started on postoperative day 17 and persisted, despite conservative treatment. Pleurodesis using oxytetracycline was done on the right side.
He was apparently well for 15 months when he was admitted to a local hospital for chest discomfort, severe breathlessness, and desaturation (PO 2, 77%). The chest discomfort was relieved after expectoration of a bronchial cast, and oxygen saturation returned to 100%. Despite treatment with nebulization, hydrocortisone, and antibiotics, he had another similar episode, after which he was referred to a tertiary care center for further management. He had a similar episode after admission to our center.
The physical examination revealed a dusky appearance. Crepitations and rhonchi were heard in the right lower zone. Apart from neutrophilic leucocytosis, the results of the other laboratory investigations were normal. The echocardiography showed a functioning fenestrated Fontan circuit with no obstruction. The Holter study was normal. Cardiac catheterization revealed a normal left ventricular end-diastolic pressure and a pressure of 13 mm of Hg in the fenestrated Fontan circuit. Bronchoscopy showed the right bronchial tree was full of casts, and the left bronchial tree was relatively normal.
Gross examination of the cast showed a whitish, rubbery cast in the shape of the bronchial tree (Fig 1). Histopathologic examination of the cast showed mucous and fibrin with enmeshed red blood cells, lymphocytes, few neutrophils, and foamy macrophages, which are indicative of a type I bronchial cast (Fig 2). Results of smears and cultures for fungi and mycobacteria were negative.
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The patient continued to expectorate bronchial casts every week, despite a low-fat diet and treatment with ß-agonist, oral anticoagulant, acetylcysteine nebulization, and corticosteroids.
When nonsurgical measures failed, thoracic duct ligation was done. Two days after the surgery, the patient suddenly desaturated. Rigid bronchoscopy showed thick secretions in the bronchi bilaterally. Repeated therapeutic bronchoscopy was done, but the saturations continued to drop and the patient died on postoperative day 8.
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Supportive care, a low-fat diet, and therapeutic bronchoscopy form the basis of palliative treatment for plastic bronchitis. Specific treatments of nebulized acetylcysteine, aerosolized urokinase, and tissue plasminogen [3] in patients refractory to conventional therapies have been tried.
Reducing pressures within the pulmonary venous circulation has resolved symptoms in some cases [1, 5]. Thoracic duct ligation has been curative in 2 patients with type II casts [7]. Five cases of type I (cellular) casts after palliative surgery for cyanotic heart disease have been reported, as was seen in our case.
Languepin and colleagues [4] reported bronchial casts containing lipids and lymphocytes in 3 patients who had immunodeficiency and pulmonary lymphatic abnormalities [4]. They propose that the pathophysiology involves the development of endobronchial lymph leakage, pulmonary lymphatic abnormalities (congenital or acquired) because of lymphatic trauma during surgery, adhesions, and elevated central venous pressure [4]. The immunodeficiency possibly leads to chronic inflammation and to the inflammatory nature of the cast.
Wilson and colleagues [5] also reported a case of plastic bronchitis with type I casts after a bidirectional Glenn shunt for single ventricle physiology where thoracic duct ligation did not help resolve the chylothorax. After the Fontan operation was completed, a second chylothorax developed that temporarily responded to a fat-free diet. The patient was readmitted for desaturation and a bronchial cast was expectorated. Thoracic duct ligation did not help resolve the chylothorax in this patient or prevent the development of plastic bronchitis. This could be because collateral channels develop between the thoracic and abdominal lymphatics in patients with persistent and recurrent chylothorax. In these patients, ligation of thoracic duct may not stop the excessive flow of lymph through the collateral channels.
Persistent leak of chyle leads to fluid and electrolyte losses and an acquired immunodeficiency from lymphocyte depletion and depletion of proteins, including immunoglobulins [8]. This acquired immunodeficiency may cause persistence of respiratory infections, resulting in bronchial hypersecretion and the formation of inflammatory bronchial cast. Thus, leakage of chyle and inflammation may both be responsible for type I cast formation. In these cases, ligation of the thoracic duct reduces the flow of chyle but does not resolve the inflammation, as was seen in our case with type I casts.
Park and colleagues [6] describe a patient with plastic bronchitis with type I casts where lobectomy resulted in complete resolution of symptoms for 4 years. It could be a treatment modality in patients with type I casts where bronchoscopy can define the main segment involved [6].
In conclusion, plastic bronchitis with a type I cast after palliative surgery for cyanotic heart disease may not benefit from thoracic duct ligation, because both leakage of chyle and inflammation may be responsible for cast formation.
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This article has been cited by other articles:
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  J. Kruger, C. Shpringer, E. Picard, and E. Kerem Thoracic Air Leakage in the Presentation of Cast Bronchitis Chest, August 1, 2009; 136(2): 615 - 617. [Abstract] [Full Text] [PDF]
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