Reports of leptospirosis have recently been increasing in Taiwan. We report a case of leptospirosis with the unusual initial manifestation of pulmonary hemorrhage. The patient presented with cough for 1 week and was admitted. After admission, fever, hemoptysis and severe dyspnea developed suddenly. Chest radiograph showed bilateral diffuse pulmonary infiltrates and he was transferred to the emergency room of our hospital. Oxygen saturation was 86% under room air and respiratory rate was 30 per minute. After admission to the thoracic ward on the third morning, parenteral penicillin and trimethoprim-sulfamethoxazole were given empirically, and a dramatic recovery ensued. Microscopic agglutination test showed an increased titer of 1:6400 against Leptospira interrogans serogroup shermani on the fourth day of hospitalization. Neither jaundice nor renal insufficiency occurred during treatment. Pulmonary hemorrhage may be an under-recognized manifestation of leptospirosis in Taiwanese patients. Leptospirosis should be taken into consideration in the differential diagnosis of pulmonary hemorrhage. Early treatment can lead to cure with reduced morbidity.