title: Altered functional brain connectivity in children and young people with opsoclonus-myoclonus syndrome.
abstract: Opsoclonus-myoclonus syndrome (OMS) is a rare, poorly understood condition that can result in long-term cognitive, behavioural, and motor sequelae. Several studies have investigated structural brain changes associated with this condition, but little is known about changes in function. This study aimed to investigate changes in brain functional connectivity in patients with OMS.
  
  
  
  Seven patients with OMS and 10 age-matched comparison participants underwent 3T magnetic resonance imaging (MRI) to acquire resting-state functional MRI data (whole-brain echo-planar images; 2mm isotropic voxels; multiband factor ×2) for a cross-sectional study. A seed-based analysis identified brain regions in which signal changes over time correlated with the cerebellum. Model-free analysis was used to determine brain networks showing altered connectivity.
  
  
  
  In patients with OMS, the motor cortex showed significantly reduced connectivity, and the occipito-parietal region significantly increased connectivity with the cerebellum relative to the comparison group. A model-free analysis also showed extensive connectivity within a visual network, including the cerebellum and basal ganglia, not present in the comparison group. No other networks showed any differences between groups.
  
  
  
  Patients with OMS showed reduced connectivity between the cerebellum and motor cortex, but increased connectivity with occipito-parietal regions. This pattern of change supports widespread brain involvement in OMS.
authors.affiliation: FMRIB Centre, Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, UK.
authors.forename: Adam M
authors.initials: AM
authors.lastname: Chekroud
authors.affiliation: Oxford Children's Hospital, John Radcliffe Hospital, Oxford, UK.
authors.forename: Geetha
authors.initials: G
authors.lastname: Anand
authors.affiliation: Oxford Children's Hospital, John Radcliffe Hospital, Oxford, UK.
authors.forename: Jean
authors.initials: J
authors.lastname: Yong
authors.affiliation: Oxford Children's Hospital, John Radcliffe Hospital, Oxford, UK.
authors.forename: Michael
authors.initials: M
authors.lastname: Pike
authors.affiliation: FMRIB Centre, Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, UK.
authors.forename: Holly
authors.initials: H
authors.lastname: Bridge
mesh_terms: D000293:Adolescent; D001921:Brain / Q000000981:diagnostic imaging* / Q000473:pathology; D001931:Brain Mapping; D016022:Case-Control Studies; D002648:Child; D003430:Cross-Sectional Studies; D005260:Female; D006801:Humans; D007091:Image Processing, Computer-Assisted; D008279:Magnetic Resonance Imaging; D008297:Male; D009434:Neural Pathways / Q000000981:diagnostic imaging*; D053578:Opsoclonus-Myoclonus Syndrome / Q000000981:diagnostic imaging* / Q000473:pathology*; D025341:Principal Component Analysis; D055815:Young Adult