Response to rituximab in 3 children with opsoclonus-myoclonus syndrome resistant to conventional treatments

Eur J Paediatr Neurol. 2012 Mar;16(2):192-5. doi: 10.1016/j.ejpn.2011.05.013. Epub 2011 Jul 6.

Abstract

We report the 1 year follow-up of 3 children affected by non-paraneoplastic Opsoclonus-Myoclonus Syndrome (OMS) resistant to conventional therapies (steroids, ACTH and intravenous immunoglobulins) who were treated with an anti CD20 monoclonal antibody (rituximab). Treatment response was recorded on the basis of an international score at 0, 3, 6, 9 and 12 months. Despite the long disease duration and the numerous previously administered treatments, all patients underwent rapid and persistent neurological recovery following rituximab administration, thus suggesting a potential role of this drug even in pre-treated patients.

Publication types

  • Case Reports

MeSH terms

  • Adrenocorticotropic Hormone / therapeutic use
  • Anti-Inflammatory Agents / therapeutic use
  • Antibodies, Monoclonal, Murine-Derived / therapeutic use*
  • Ataxia / drug therapy
  • Ataxia / etiology
  • Child Behavior Disorders / etiology
  • Child Behavior Disorders / psychology
  • Dexamethasone / therapeutic use
  • Drug Resistance
  • Electroencephalography
  • Gait Disorders, Neurologic / drug therapy
  • Gait Disorders, Neurologic / etiology
  • Humans
  • Immunoglobulins, Intravenous / therapeutic use
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Opsoclonus-Myoclonus Syndrome / drug therapy*
  • Prednisone / therapeutic use
  • Rituximab
  • Speech Disorders / drug therapy
  • Speech Disorders / etiology
  • Treatment Outcome
  • Tremor / drug therapy
  • Tremor / etiology
  • Virus Diseases / complications

Substances

  • Anti-Inflammatory Agents
  • Antibodies, Monoclonal, Murine-Derived
  • Immunoglobulins, Intravenous
  • Rituximab
  • Dexamethasone
  • Adrenocorticotropic Hormone
  • Prednisone