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Lymphangiomatosis presenting with bronchial cast formation
1Department of Pulmonary and Critical Care Medicine, Norwalk Hospital, Connecticut 06850, USA.
Thorax
|July 1, 1996
Summary
Lymphangiomatosis, a lymphatic system disorder, can cause chylothorax and rare chylous bronchial casts. Surgical thoracic duct ligation successfully treated a patient presenting with bronchial casts and subsequent chylothorax.
Area of Science:
- Medicine
- Pulmonology
- Vascular Medicine
Background:
- Lymphangiomatosis is a rare congenital malformation of the lymphatic system.
- It can affect various organs, leading to diverse clinical manifestations.
- Chylothorax, the accumulation of chylous fluid in the pleural space, is a known complication.
Observation:
- A 34-year-old woman presented with a rare initial symptom of lymphangiomatosis: cough productive of bronchial casts.
- Chyloptysis (expectoration of chylous fluid) was not reported in this case.
- Two years after the initial presentation, the patient developed chylothorax.
Findings:
- Chylous bronchial cast formation is an exceptionally rare manifestation of lymphangiomatosis.
- The development of chylothorax followed the bronchial cast symptom by two years.
- Successful treatment was achieved through ligation of the thoracic duct.
Implications:
- This case highlights the varied and sometimes unusual presentations of lymphangiomatosis.
- It underscores the importance of considering rare diagnoses in patients with unexplained respiratory symptoms and effusions.
- Thoracic duct ligation is a viable and potentially curative surgical option for lymphangiomatosis-induced chylothorax.