Besides the classical manifestations, leptospirosis may rarely occur with erythroid hypoplasia and/or pancytopenia. In this study, we reported two cases of leptospirosis presented with pancytopenia as the prevailing manifestation. In addition, the presence of pancytopenia in leptospirosis is reviewed. In both patients, the outcome was favourable as the bone marrow aplasia reversed completely after treatment with intravenous penicillin. In conclusion, this case study suggests that Leptospira infection should be included in the differential diagnosis of febrile pancytopenia, even in the absence of classical signs of severe disease as jaundice, meningitis, renal failure and pulmonary infiltrates.