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Churg-Strauss syndrome (CSS) is a rare multi-system vasculitis. Patients with CSS are at greater risk of venous thromboembolic events. However, venous thrombosis is rarely reported in patients with CSS in Korea. We report 1 patient with CSS who experienced deep vein thrombosis. A 27- year-old man was admitted to our hospital due to fever, cough, skin rash on both lower extremities, and right leg edema. He was diagnosed with CSS based on a history of asthma, peripheral eosinophilia, transient pulmonary infiltrates on chest radiograph, and extravascular eosinophils in the skin biopsy specimens. After steroid pulse therapy and the first cycle of intravenous pulse cyclophosphamide therapy, right leg edema did not resolve. Venous ultrasonography and CT angiography detected deep vein thrombosis involving the right common iliac, femoral and calf veins. The patient received both anticoagulation therapy and intravenous pulse cyclophosphamide therapy. After 6 months of anticoagulation therapy and the 9 cycles of intravenous pulse cyclophosphamide therapy, deep vein thrombosis completely resolved.

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