title: Rituximab treatment for relapsed opsoclonus-myoclonus syndrome.
abstract: BACKGROUND
  Opsoclonus-myoclonus syndrome (OMS) is a rare neurological disorder that is associated with paraneoplastic diseases. Because OMS can frequently relapse, patients may be inflicted with neurological problems for a long time. Recently, rituximab (RTX) was introduced as a drug to treat OMS. To assess RTX treatment, we studied a patient who experienced recurrence of OMS.
  
  
  METHODS
  A 2-year-old Japanese boy, who had left adrenal neuroblastoma, suddenly showed OMS symptoms, including ataxia and opsoclonus. Surgical resection of the tumor and subsequent steroid therapy ameliorated his symptoms. When OMS relapsed during the time when prednisolone was reduced, he was treated with full-dose RTX therapy (375 mg/m2/week) for 4 consecutive weeks. However, 1year later, he presented again with OMS symptoms. This time, we only administered an additional single dose of RTX treatment (375 mg/m2), allowing remission of OMS symptoms. During 2 years after the additional RTX treatment, OMS symptoms did not appear, even when prednisolone was reduced. He had no adverse events associated with RTX during the whole treatment period.
  
  
  CONCLUSIONS
  An additional single-dose RTX therapy might be effective for relapsed OMS patients who were previously treated with full-dose RTX therapy.
authors.affiliation: Department of Pediatrics, Kobe University Graduate School of Medicine, Japan.
authors.forename: Daisaku
authors.initials: D
authors.lastname: Toyoshima
authors.affiliation: Department of Pediatrics, Kobe University Graduate School of Medicine, Japan. Electronic address: morisada@med.kobe-u.ac.jp.
authors.forename: Naoya
authors.initials: N
authors.lastname: Morisada
authors.affiliation: Department of Pediatrics, Himeji Red Cross Hospital, Japan.
authors.forename: Yuichi
authors.initials: Y
authors.lastname: Takami
authors.affiliation: Department of Pediatrics, Nagoya University Graduate School of Medicine, Japan.
authors.forename: Hiroyuki
authors.initials: H
authors.lastname: Kidokoro
authors.affiliation: Department of Pediatrics, Kobe University Graduate School of Medicine, Japan.
authors.forename: Masahiro
authors.initials: M
authors.lastname: Nishiyama
authors.affiliation: Department of Pediatrics, Himeji Red Cross Hospital, Japan.
authors.forename: Taku
authors.initials: T
authors.lastname: Nakagawa
authors.affiliation: Department of Pediatrics, Kobe University Graduate School of Medicine, Japan.
authors.forename: Takeshi
authors.initials: T
authors.lastname: Ninchoji
authors.affiliation: Department of Pediatrics, Kobe University Graduate School of Medicine, Japan.
authors.forename: Kandai
authors.initials: K
authors.lastname: Nozu
authors.affiliation: Department of Pediatrics, Hyogo College of Medicine, Japan.
authors.forename: Yasuhiro
authors.initials: Y
authors.lastname: Takeshima
authors.affiliation: Kobe University Graduate School of Health Science, Japan.
authors.forename: Satoshi
authors.initials: S
authors.lastname: Takada
authors.affiliation: Division of Epidemiology, Kobe University Graduate School of Medicine, Japan.
authors.forename: Hisahide
authors.initials: H
authors.lastname: Nishio
authors.affiliation: Department of Pediatrics, Kobe University Graduate School of Medicine, Japan.
authors.forename: Kazumoto
authors.initials: K
authors.lastname: Iijima
mesh_terms: D002675:Child, Preschool; D006801:Humans; D008297:Male; D009364:Neoplasm Recurrence, Local / Q000188:drug therapy; D009447:Neuroblastoma / Q000503:physiopathology / Q000601:surgery; D053578:Opsoclonus-Myoclonus Syndrome / Q000188:drug therapy*; D011239:Prednisolone / Q000627:therapeutic use; D000069283:Rituximab / Q000627:therapeutic use*
chemical_list: D000069283:Rituximab; D011239:Prednisolone
keywords: Neuroblastoma; Opsoclonus–myoclonus syndrome; Rituximab; Single dose