title: Severe inflammatory disease activity 14 months after cessation of Natalizumab in a patient with Leber's optic neuropathy and multiple sclerosis - a case report.
abstract: BACKGROUND
  Leber's hereditary optic neuropathy (LHON) co-occuring with multiple sclerosis-like disease (LHON-MS) is suggested to be a separate disease entity denoted Harding's disease. Little is known about the response to initiation and discontinuation of potent immunomodulatory treatment in LHON-MS.
  
  
  METHODS
  We describe a LHON-MS patient with 27 years disease duration who developed severe disease activity peaking 14 months after discontinuation of natalizumab, with extensive new inflammatory lesions throughout the brain and in the spinal cord resembling immune inflammatory reconstitution syndrome. She had previously been clinically and radiologically stable on natalizumab treatment for 6 years, and before that only experienced subtle clinical activity during 9 years on interferon beta1a.
  
  
  CONCLUSIONS
  This is the first report on severe exacerbation of inflammatory disease activity after discontinuation of natalizumab in LHON-MS, and suggests that late rebound activity can occur in these patients.
authors.affiliation: Department of Neurology, Akershus University Hospital, Lørenskog, Norway. trygve.holmoy@medisin.uio.no.|Institute of Clinical Medicine, University of Oslo, Oslo, Norway. trygve.holmoy@medisin.uio.no.
authors.forename: Trygve
authors.identifier: http://orcid.org/0000-0002-2529-5664
authors.initials: T
authors.lastname: Holmøy
authors.affiliation: Multiple Sclerosis Centre Hakadal, Hakadal, Norway.
authors.forename: Antonie G
authors.initials: AG
authors.lastname: Beiske
authors.affiliation: Department of Radiology, Akershus University Hospital, Lørenskog, Norway.
authors.forename: Svetozar
authors.initials: S
authors.lastname: Zarnovicky
authors.affiliation: Department of Neurology, Akershus University Hospital, Lørenskog, Norway.
authors.forename: Aija Zuleron
authors.initials: AZ
authors.lastname: Myro
authors.affiliation: Department of Neurology, Akershus University Hospital, Lørenskog, Norway.|Institute of Clinical Medicine, University of Oslo, Oslo, Norway.
authors.forename: Egil
authors.initials: E
authors.lastname: Røsjø
authors.affiliation: Institute of Clinical Medicine, University of Oslo, Oslo, Norway.|Department of Neurology, Oslo University Hospital Rikshospitalet, Oslo, Norway.
authors.forename: Emilia
authors.initials: E
authors.lastname: Kerty
mesh_terms: D000328:Adult; D001921:Brain / Q000473:pathology; D005260:Female; D006801:Humans; D007249:Inflammation / Q000150:complications* / Q000000981:diagnostic imaging / Q000473:pathology; D008279:Magnetic Resonance Imaging; D008297:Male; D009103:Multiple Sclerosis / Q000150:complications* / Q000000981:diagnostic imaging / Q000188:drug therapy / Q000473:pathology*; D000069442:Natalizumab / Q000008:administration & dosage* / Q000627:therapeutic use*; D029242:Optic Atrophy, Hereditary, Leber / Q000150:complications* / Q000000981:diagnostic imaging / Q000188:drug therapy / Q000473:pathology*; D012008:Recurrence; D013116:Spinal Cord / Q000473:pathology; D013997:Time Factors
chemical_list: D000069442:Natalizumab
keywords: Case report; Immune reconstitution inflammatory syndrome; Leber’s optic neuropathy; Multiple sclerosis; Natalizumab; Rebound