title: Cerebrospinal fluid oxidative stress marker levels and cytokine concentrations in a neonate with incontinentia pigmenti.
abstract: BACKGROUND
  Some children with incontinentia pigmenti exhibit encephalopathic features with severe seizures and disturbed consciousness, from the neonatal through the early infantile period. However, the pathological mechanism of brain lesion development is not fully understood.
  
  
  METHODS
  We measured the cerebrospinal fluid levels of cytokines and oxidative stress markers (8-hydroxy-2-deoxyguanosine and the hexanoyl-lysine adduct) in a young girl with incontinentia pigmenti complicated by an encephalopathic event that occurred on her first day of life. Magnetic resonance imaging revealed widespread reduction of water diffusion in the basal ganglia, the periventricular and subcortical white matter, and the corpus callosum.
  
  
  RESULTS
  Oxidative stress markers were elevated at 4 days of age but decreased mildly by 25 days of age. Elevated levels of soluble tumor necrosis factor receptor 1 were observed at both 4 and 25 days of age, although tumor necrosis factor-α levels were below the limit of detection. No other cytokine levels were elevated, except for those of interleukin-10 at 25 days of age.
  
  
  CONCLUSIONS
  Tumor necrosis factor-α expression and oxidative stress are involved in the pathogenesis of brain lesions in children with incontinentia pigmenti, and elevated cerebrospinal fluid cytokine levels may not be apparent during encephalopathic events.
authors.affiliation: Neonatal Intensive Care Unit, Juntendo Shizuoka Hospital, Izunokuni, Japan.
authors.forename: Natsuki
authors.initials: N
authors.lastname: Ohkawa
authors.affiliation: Department of Pediatrics, Faculty of Medicine, Juntendo University, Tokyo, Japan; Department of Pediatrics, Aichi Medical University, Nagakute, Japan. Electronic address: okumura.akihisa.479@mail.aichi-med-u.ac.jp.
authors.forename: Akihisa
authors.initials: A
authors.lastname: Okumura
authors.affiliation: Department of Brain Development and Neural Regeneration, Tokyo Metropolitan Institute of Medical Science, Tokyo, Japan.
authors.forename: Rie
authors.initials: R
authors.lastname: Miyata
authors.affiliation: Department of Brain Development and Neural Regeneration, Tokyo Metropolitan Institute of Medical Science, Tokyo, Japan.
authors.forename: Naoyuki
authors.initials: N
authors.lastname: Tanuma
authors.affiliation: Department of Brain Development and Neural Regeneration, Tokyo Metropolitan Institute of Medical Science, Tokyo, Japan.
authors.forename: Masaharu
authors.initials: M
authors.lastname: Hayashi
authors.affiliation: Neonatal Intensive Care Unit, Juntendo Shizuoka Hospital, Izunokuni, Japan.
authors.forename: Hiroaki
authors.initials: H
authors.lastname: Sato
authors.affiliation: Department of Pediatrics, Faculty of Medicine, Juntendo University, Tokyo, Japan.
authors.forename: Toshiaki
authors.initials: T
authors.lastname: Shimizu
mesh_terms: D000080242:8-Hydroxy-2'-Deoxyguanosine; D015415:Biomarkers; D016207:Cytokines / Q000134:cerebrospinal fluid*; D003849:Deoxyguanosine / Q000031:analogs & derivatives / Q000134:cerebrospinal fluid; D004797:Enzyme-Linked Immunosorbent Assay; D005260:Female; D006801:Humans; D007184:Incontinentia Pigmenti / Q000134:cerebrospinal fluid* / Q000473:pathology; D007231:Infant, Newborn; D008239:Lysine / Q000134:cerebrospinal fluid*; D008279:Magnetic Resonance Imaging; D018384:Oxidative Stress / Q000502:physiology
chemical_list: D015415:Biomarkers; D016207:Cytokines; D000080242:8-Hydroxy-2'-Deoxyguanosine; D003849:Deoxyguanosine; D008239:Lysine
keywords: 8-hydroxy-2-deoxyguanosine; cytokines; hexanoyl-lysine adduct; incontinentia pigmenti; soluble tumor necrosis factor receptor 1