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Background A growing body of evidence, in animals and humans, suggest that IFNg plays a pathogenic role in primary HLH (pHLH) and in secondary HLH, including macrophage activation syndrome. A pilot study in pHLH with NI-0501, an anti-IFNg monoclonal antibody, is ongoing. Mutations in NLRC4 have been recently reported to cause recurrent MAS and an increased production of IL-18, a cytokine known to induce IFNg.

Anti interferon-gamma (IFNγ) monoclonal antibody treatment in a patient carrying an NLRC4 mutation and severe hemophagocytic lymphohistiocytosis