Success of anti-CD20 monoclonal antibody treatment for severe autoimmune hemolytic anemia caused by warm-reactive immunoglobulin A, immunoglobulin G, and immunoglobulin M autoantibodies in a child: a case report

Abstract

BackgroundAutoimmune hemolytic anemia is rare in children. First-line therapies for this disease consist of corticosteroids and intravenously administered immunoglobulin that are effective in most patients. However, a small proportion of cases (5 to 10%) is refractory to these therapies and may represent a medical emergency, especially when hemolysis is due to warm immunoglobulin M. Recently, reports of the use of rituximab in adult autoimmune diseases have shown promising results. In children, there are few studies on the use of rituximab in the treatment for autoimmune hemolytic anemia, especially on its long-term efficacy and adverse effects.Case presentationHere, we report the case of a 10-year-old Tunisian girl with refractory acute autoimmune hemolytic anemia caused by warm-reactive immunoglobulin A, immunoglobulin G, immunoglobulin M, and C3d autoantibodies. First-line treatments using corticosteroids and intravenously administered immunoglobulin were ineffective in controlling her severe disease. On the other hand, she was successfully treated with rituximab. In fact, her hemolytic anemia improved rapidly and no adverse effects were observed.ConclusionsThe case that we report in this paper shows that rituximab could be an alternative therapeutic option in severe acute autoimmune hemolytic anemia with profound hemolysis refractory to conventional treatment. Moreover, it may preclude the use of plasmapheresis in such an urgent situation with a sustained remission.

IgM-mediated Warm Autoimmune Hemolytic Anemia: An Autopsy Report

“…In previous case reports, treatment with steroids and/or transfusion was mainly performed in patients with IgM-mediated warm AIHA. However, in several cases, high-dose intravenous immunoglobulin and plasma exchange therapies were provided to the patients, or rituximab was administered (7,11). Recently, a randomized and double-blind controlled trial evaluating the safety and efficacy of rituximab for warm AIHA in adults (the RAIHA study) showed that the administration of rituximab combined with steroids for newly developed warm AIHA in adults significantly improved the complete remission and mortality rates compared to placebo (12).…”

Correlation between serum immunoglobulin levels and retinal structure in patients with newly diagnosed Vogt‑Koyanagi‑Harada disease

“…Igs also serve immunomodulatory roles by neutralizing specific antigens and activating the complement system ( 5 ). Studies have demonstrated that serum IgG, IgA and IgM also serve important roles in numerous diseases, including autoimmune hemolytic anemia, IgA nephropathy and autoimmune hepatitis ( 6 – 8 ). It has also been reported that the serum IgG, IgA and IgM levels were significantly increased in patients with rheumatoid arthritis and the changes in these levels were associated with disease activity ( 9 ).…”

Anti-CD20 Treatment of Autoimmune Hemolytic Anemia Refractory to Corticosteroids and Azathioprine: A Pediatric Case Report and Mini Review

“…In adults, a recent meta-analysis from several studies showed that the overall response rate and complete response rate were nearly 70% and 40%, respectively, for warm AIHA [ 9 ]. In children, the majority of the available publications are case reports or single-center uncontrolled studies [ 4 , 7 , 8 , 10 – 14 ]. In a recent prospective national French study of 61 children, the 6-year relapse-free survival was 48% [ 15 ].…”