Background: B cell depletion with rituximab, a genetically engineered chimeric anti-CD20 monoclonal antibody has been shown to be effective in certain chronic immunological diseases including rheumatoid arthritis, systemic lupus erythematosus, and antineutrophil cytoplasmic antibody (ANCA) associated vasculitis. Several published reports have shown its effectiveness in Churg-Strauss Syndrome. Case report: We report a case of a 56 year old Filipino male, with a prior history of asthma and sinus abnormalities, who presented with peripheral eosinophilia, leukocytoclastic vasculitis, and severe axonal distal symmetrical sensorimotor polyneuropathy. Sural nerve biopsy revealed vasculitic neuropathy. He had received intravenous immunoglobulin, methylprednisolone pulses and high dose oral steroids but without clinical improvement. He was then given 2 doses of rituximab 500 mg combined with 500 mg cyclophosphamide, administered 2 weeks apart, without adverse events. A month later, his neurologic functions had significantly improved with increasing ambulation, and prednisone was effectively tapered to 10 mg daily. Conclusion: Rituximab is an important option in the management of systemic vasculitis including Churg-Strauss syndrome refractory to the usual immunosuppressive agents.