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Waldenström macroglobulinemia is a lymphoplasmocytic lymphoma involving bone marrow and characterized by the production of a monoclonal IgM gammapathy. Despite conventional chemotherapies it is an incurable disease. The recent discovery of the pro-inflammatory transcription factor MYD88 L265P mutation as a molecular signature of the disease could represent a new therapeutic target. We report here, a case of Waldenström macroglobulinemia associated with a high biological inflammatory syndrome, resistant to conventional therapies and improved by tocilizumab, an anti-interleukin-6 Receptor inhibitor.