title: Successful treatment of autoimmune hemolytic anemia associated with multicentric Castleman disease by anti-interleukin-6 receptor antibody (tocilizumab) therapy.
abstract: We describe herein the successful treatment of severe autoimmune hemolytic anemia (AIHA) in a patient with multicentric Castleman disease (MCD) by humanized anti-interleukin-6 (IL-6) receptor antibody (tocilizumab) therapy. Inflammatory anemia is commonly reported; however, AIHA is a very rare complication of MCD. In 1996, a 45-year-old Japanese woman was referred to our hospital because of generalized lymphadenopathy, anemia and skin eruptions. Lymph node biopsy demonstrated MCD. She was treated with prednisolone (1 mg/kg/day), which improved the anemia and skin eruptions. In 2009, she suddenly developed Coombs-positive hemolytic anemia. The blood count was as follows: hemoglobin 4.7 g/dl, platelets 490 × 10(9)/l and white blood cell count 9.8 × 10(9)/l. Both direct and indirect Coombs' tests were strongly positive. She was treated with 8 mg/kg tocilizumab every 2 weeks. One month later, her hemoglobin levels rose dramatically to 10.9 g/dl and her haptoglobin level, hypergammaglobulinemia and clinical symptoms had also markedly improved. To the best of our knowledge, this is the first report of the efficacy of tocilizumab in AIHA associated with MCD. The well-established role of IL-6 in the pathogenesis of MCD may have been responsible for the improvement in the AIHA associated with MCD. Anti-IL-6 receptor antibody treatment could be an attractive therapeutic approach for AIHA associated with MCD.
authors.affiliation: Department of Medicine and Clinical Science, Gunma University Graduate School of Medicine, Maebashi, Japan.
authors.forename: Akinori
authors.initials: A
authors.lastname: Yuzuriha
authors.forename: Takayuki
authors.initials: T
authors.lastname: Saitoh
authors.forename: Hiromi
authors.initials: H
authors.lastname: Koiso
authors.forename: Takeki
authors.initials: T
authors.lastname: Mitsui
authors.forename: Hideki
authors.initials: H
authors.lastname: Uchiumi
authors.forename: Akihiko
authors.initials: A
authors.lastname: Yokohama
authors.forename: Hiroshi
authors.initials: H
authors.lastname: Handa
authors.forename: Masaru
authors.initials: M
authors.lastname: Kojima
authors.forename: Norifumi
authors.initials: N
authors.lastname: Tsukamoto
authors.forename: Masamitsu
authors.initials: M
authors.lastname: Karaswa
authors.forename: Hirokazu
authors.initials: H
authors.lastname: Murakami
authors.forename: Yoshihisa
authors.initials: Y
authors.lastname: Nojima
mesh_terms: D000744:Anemia, Hemolytic, Autoimmune / Q000150:complications / Q000188:drug therapy*; D000911:Antibodies, Monoclonal / Q000276:immunology / Q000627:therapeutic use*; D061067:Antibodies, Monoclonal, Humanized; D005871:Castleman Disease / Q000150:complications*; D005260:Female; D006801:Humans; D008875:Middle Aged; D019947:Receptors, Interleukin-6 / Q000276:immunology*; D016896:Treatment Outcome
chemical_list: D000911:Antibodies, Monoclonal; D061067:Antibodies, Monoclonal, Humanized; D019947:Receptors, Interleukin-6; C502936:tocilizumab