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Background: Acquired haemophilia is an uncommon condition caused by the development of clotting factor inhibitors. To eliminate them, immunosuppressive therapy with corticosteroids and cytotoxic drugs is required. Methods: We describe a case of rituximab use in acquired haemophilia refractory to conventional therapy in a 63 year old male patient with chronic hepatitis C virus infection who was receiving treatment with pegylated-interferon-α-2a plus ribavirin. Results: After 21 weeks of antiviral therapy, the patient was admitted to hospital with a large haematoma in the abdominal muscles. Factor VIII level was zero and inhibitor titer was 345 Bethesda units. Oral immunosuppressive therapy with methylprednisolone and cyclophosphamide was administered for 1 month, with limited improvement. Therefore, cyclophosphamide was replaced by a four once-weekly dose of intravenous rituximab. Two months later, factor VIII level was normal and inhibitor titer was undetectable. Conclusion: Rituximab may be useful for the treatment of acquired haemophilia resistant to standard therapy.