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Lymphoscintigraphy in plastic bronchitis, a pediatric case report
Dima R Ezmigna1, Wayne J Morgan, Marlys H Witte
1Department of Pediatrics, University of Arizona, Tucson, Arizona 85724, USA. dezmigna@yahoo.com
Insights
Plastic bronchitis involves bronchial cast formation. In a patient with congenital heart disease, lymphoscintigraphy revealed lymphatic leakage contributing to cast formation, suggesting a potential treatment target.
Area of Science:
- Pulmonary Medicine
- Cardiology
- Medical Imaging
Background:
- Plastic bronchitis (PB) is a rare, severe condition characterized by bronchial cast formation.
- Underlying causes often involve pulmonary lymphatic abnormalities.
- Congenital heart disease (CHD), particularly post-Fontan procedure, is a risk factor.
Observation:
- A 6-year-old male with CHD presented with recurrent respiratory issues.
- Initial bronchoscopy showed inflammation and mucus, but no casts.
- PB was diagnosed after the patient expectorated a bronchial cast.
Findings:
- Cast analysis revealed lymphocytic aggregates, mucin, and fibrin.
- Lymphoscintigraphy demonstrated abnormal lymphatic collaterals and retrograde lymph reflux.
- This indicated thoracic duct lymph leakage into the tracheobronchial tree.
Implications:
- High intrathoracic lymphatic pressure and retrograde flow may cause recurrent cast formation in post-Fontan patients.
- Identifying lymphatic defects is crucial for managing PB.
- Lymphoscintigraphy offers a safer alternative to lymphangiography for diagnosing lymphatic abnormalities.
Abstract:
Plastic bronchitis (PB) is an uncommon, potentially fatal disease, marked by endobronchial cast formation causing variable degrees of respiratory distress. Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6-year-old Hispanic male with congenital heart disease (CHD; post-Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic-venous shunting may be possible in select cases.
