Dian He1, YunLi Yu1, WeiBo Yan1, QingQing Dai1, Zhu Xu1, Lan Chu2. 1. Department of Neurology, Affiliated Hospital of Guiyang Medical College, Guiyang, Guizhou Province, China. 2. Department of Neurology, Affiliated Hospital of Guiyang Medical College, Guiyang, Guizhou Province, China. Electronic address: chulan8999@sohu.com.
Abstract
BACKGROUND: Neuromyelitis optica is an autoimmune inflammatory disorder of the central nervous system. Current therapeutic approaches are based on small uncontrolled trials, case series, or case reports. There are only a few case reports describing rituximab for pediatric neuromyelitis optica. PATIENT: A 7-year-old girl with neuromyelitis optica had high disease activity with recurrent myelitis and steroid dependence. A remarkable increase of CD19(+) B-cell count in the peripheral blood mononuclear cells and seropositivity for anti-aquaporin 4 antibody were detected at each attack. After induction therapy with rituximab, the CD19(+) B-cell number was significantly reduced and sustained at low levels. The level of serum anti-aquaporin 4 antibody normalized. She was relapse-free over 1-year follow-up period. An individualized maintenance therapy scheme is underway. CONCLUSION: Treatment with rituximab for relapsing neuromyelitis optica requires an individualized regimen to optimize the frequency and dosage of administration to maximize efficacy yet minimize overtreatment and cost. Personal levels of CD19(+) B cells in peripheral blood mononuclear cells at previous attacks and responsiveness to rituximab in induction therapy may be two useful indicators in establishing individualized maintenance therapy schemes for relapsing neuromyelitis optica.
BACKGROUND:Neuromyelitis optica is an autoimmune inflammatory disorder of the central nervous system. Current therapeutic approaches are based on small uncontrolled trials, case series, or case reports. There are only a few case reports describing rituximab for pediatric neuromyelitis optica. PATIENT: A 7-year-old girl with neuromyelitis optica had high disease activity with recurrent myelitis and steroid dependence. A remarkable increase of CD19(+) B-cell count in the peripheral blood mononuclear cells and seropositivity for anti-aquaporin 4 antibody were detected at each attack. After induction therapy with rituximab, the CD19(+) B-cell number was significantly reduced and sustained at low levels. The level of serum anti-aquaporin 4 antibody normalized. She was relapse-free over 1-year follow-up period. An individualized maintenance therapy scheme is underway. CONCLUSION: Treatment with rituximab for relapsing neuromyelitis optica requires an individualized regimen to optimize the frequency and dosage of administration to maximize efficacy yet minimize overtreatment and cost. Personal levels of CD19(+) B cells in peripheral blood mononuclear cells at previous attacks and responsiveness to rituximab in induction therapy may be two useful indicators in establishing individualized maintenance therapy schemes for relapsing neuromyelitis optica.
Authors: Margherita Nosadini; Gulay Alper; Catherine J Riney; Leslie A Benson; Shekeeb S Mohammad; Sudarshini Ramanathan; Melinda Nolan; Richard Appleton; Richard J Leventer; Kumaran Deiva; Fabienne Brilot; Mark P Gorman; Amy T Waldman; Brenda Banwell; Russell C Dale Journal: Neurol Neuroimmunol Neuroinflamm Date: 2016-01-21