Abstract
Gorham's disease (GD) is rare and characterized by non-neoplastic lymphovascular proliferation and massive osteolysis. Its clinical course is usually protracted, but sometimes life threatening when vital structures are involved or when complicated with chylothorax. There is no optimal treatment guideline for GD complicated with chylothorax. Surgical ligation of thoracic duct, pleurectomy, pleurodesis, interferon-alpha-2b, and radiotherapy, are reported to manage chylothorax. We present the case of a 32-year-old man with Gorham's disease complicated by bilateral chylothorax, which was refractory to radiotherapy but remitted dramatically two weeks after interferon-alpha-2a therapy. The patient was free of relapse four months after discontinuing four-month interferon therapy. To date, only 11 cases (including ours) of GD with chylothorax have received interferon as single or salvage therapy, and 10 of them survived. Early intervention with interferon therapy can be considered as an effective treatment for GD complicated with bilateral chylothorax.
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