Paradoxical reactions and immune deviation following biologic therapy are occasionally observed in clinical practice; however, to our knowledge, no previous cases of paradoxical skin rash accompanied by Hyperimmunoglobulinemia E have been reported.<h4>Case presentation</h4>A 35-year-old male with SAPHO syndrome experienced significant exacerbation of palmoplantar pustulosis and a sharp increase in immunoglobulin E(IgE) levels following treatment with secukinumab and infliximab. This evidence concerns the gene IGHE and SAPHO syndrome.