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High association of MOG-IgG antibodies in children with bilateral optic neuritis.
Wendel, Eva-Maria; Baumann, Matthias; Barisic, Nina; Blaschek, Astrid; Coelho de Oliveira Koch, Eliana; Della Marina, Adela; Diepold, Katharina; Hackenberg, Annette; Hahn, Andreas; von Kalle, Thekla; Karenfort, Michael; Kornek, Barbara; Lechner, Christian; Leiz, Steffen; Merkenschlager, Andreas; Nosadini, Margherita; Sartori, Stefano; Schanda, Kathrin; Schimmel, Mareike; Seemann, Larissa; Tüngler, Victoria; Waltz, Stephan; Wegener-Panzer, Andreas; Wiegand, Gert; Reindl, Markus; Rostásy, Kevin.
Afiliação
  • Wendel EM; Department of Pediatrics, Olgahospital, Klinikum, Stuttgart, Germany.
  • Baumann M; Division of Pediatric Neurology, Department of Pediatrics I, Medical University of Innsbruck, Austria.
  • Barisic N; Department of Pediatrics, Clinical Medical Center Zagreb, University of Zagreb Medical School, Croatia.
  • Blaschek A; Department of Pediatric Neurology, Dr. von Hauner Children's Hospital, Ludwig-Maximillian-Universität Munich, Germany.
  • Coelho de Oliveira Koch E; Division of Pediatric Neurology, Department of Pediatrics, Medical University of Geneva, Switzerland.
  • Della Marina A; Department of Neuropediatrics, Developmental Neurology and Social Pediatrics, Children's Hospital, University of Duisburg-Essen, Germany.
  • Diepold K; Division of Pediatric Neurology, Department of Pediatrics, Hospital Kassel, Germany.
  • Hackenberg A; Department of Pediatric Neurology, University Children's Hospital, Zürich, Switzerland.
  • Hahn A; Division of Pediatric Neurology, Department of Pediatrics, Medical University Giessen, Germany.
  • von Kalle T; Department of Pediatric Radiology, Olgahospital, Klinikum Stuttgart, Germany.
  • Karenfort M; Department of Pediatrics, Neonatology and Pediatric Cardiology, Children's Hospital, Heinrich-Heine-University, Düsseldorf, Germany.
  • Kornek B; Department of Neurology, Medical University Vienna, Austria.
  • Lechner C; Division of Pediatric Neurology, Department of Pediatrics I, Medical University of Innsbruck, Austria.
  • Leiz S; Division of Pediatric Neurology, Department of Pediatrics, Klinikum Dritter Orden, Munich, Germany.
  • Merkenschlager A; Division of Pediatric Neurology, Department of Pediatrics, Medical University of Leipzig, Germany.
  • Nosadini M; Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Italy.
  • Sartori S; Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Italy.
  • Schanda K; Clinical Department of Neurology, Medical University of Innsbruck, Austria.
  • Schimmel M; Division of Pediatric Neurology, Children's Hospital, Medical University of Augsburg, Germany.
  • Seemann L; Department of Pediatric Neurology, Children's Hospital DRK Siegen, Germany.
  • Tüngler V; Division of Pediatric Neurology, Department of Pediatrics, Medical University Carl Gustav Carus, Dresden, Germany.
  • Waltz S; Department of Pediatric Neurology, Children's Hospital Amsterdamer Straße, Cologne, Germany.
  • Wegener-Panzer A; Department of Pediatric Radiology, Children's Hospital Datteln, University Witten/Herdecke, Germany.
  • Wiegand G; Division of Pediatric Neurology, Department of Pediatrics, Asklepios Klinik Nord, Heidberg/Hamburg, Germany.
  • Reindl M; Clinical Department of Neurology, Medical University of Innsbruck, Austria.
  • Rostásy K; Department of Pediatric Neurology, Children's Hospital Datteln, University Witten/Herdecke, Germany. Electronic address: k.rostasy@kinderklinik-datteln.de.
Eur J Paediatr Neurol ; 27: 86-93, 2020 Jul.
Article em En | MEDLINE | ID: mdl-32327391
ABSTRACT

BACKGROUND:

Bilateral optic neuritis (bilON) is a rare clinical presentation often thought to be associated with relapsing disorders such as neuromyelitis optica spectrum disorders (NMOSD) or multiple sclerosis (MS).

OBJECTIVE:

To characterize the clinical, radiological phenotype and antibody status of children presenting with bilON. MATERIAL AND

METHODS:

Retrospective multicenter study on children with bilON age <18 years with a first episode aquired demyelinating syndrome (ADS), cMRI, AQP4- and serum MOG-antibody status and follow-up data were collected.

RESULTS:

30 patients (fm = 1515, median age 8.0y) with bilON met the inclusion criteria. 22/30 (73%) were MOG-positive (median 11280, range 1160-11520). No patient showed AQP4-abs. 4/30 patients (13%), all with high MOG-abs titers, had recurrent episodes. No patient developed MS. Improvement after IVMP was observed in most patients (26/30; 87%). Outcome was favorable with no sequelae in 22/30 patients. Serial MOG-abs titers tested in 15/22 patients decreased to a median of 1160 (range 0-1640) over a period of 31 months (range 2-141 months) in 14/15 (93%) patients. MR imaging showed a predominantly anterior affection of the visual system in seropositive patients with bilateral intraorbital lesions in 68% (15/22), compared to 25% in MOG-negative patients (2/8).

CONCLUSION:

Pediatric bilON is associated with high MOG-abs titers in combination with anterior involvement of the visual system. Despite severe loss of vision, the majority of patients shows distinct recovery after IVMP.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Autoanticorpos / Neurite Óptica / Proteína ran de Ligação ao GTP Tipo de estudo: Observational_studies / Prognostic_studies / Risk_factors_studies Limite: Adolescent / Child / Child, preschool / Female / Humans / Male Idioma: En Revista: Eur J Paediatr Neurol Assunto da revista: NEUROLOGIA / PEDIATRIA Ano de publicação: 2020 Tipo de documento: Article País de afiliação: Alemanha

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Autoanticorpos / Neurite Óptica / Proteína ran de Ligação ao GTP Tipo de estudo: Observational_studies / Prognostic_studies / Risk_factors_studies Limite: Adolescent / Child / Child, preschool / Female / Humans / Male Idioma: En Revista: Eur J Paediatr Neurol Assunto da revista: NEUROLOGIA / PEDIATRIA Ano de publicação: 2020 Tipo de documento: Article País de afiliação: Alemanha