Pediatric optic neuritis and anti MOG antibodies: a cohort of Italian patients.

Acquired demyelinating syndromes of the central nervous system Myelin oligodendrocyte glycoprotein Optic disc swelling Spectral domain optical coherence tomography VEP abnormalities

Journal

Multiple sclerosis and related disorders
ISSN: 2211-0356
Titre abrégé: Mult Scler Relat Disord
Pays: Netherlands
ID NLM: 101580247

Informations de publication

Date de publication:
Apr 2020
Historique:
received: 23 08 2019
revised: 16 12 2019
accepted: 23 12 2019
medline: 3 1 2020
pubmed: 3 1 2020
entrez: 3 1 2020
Statut: ppublish

Résumé

recent studies reported that anti myelin oligodendrocyte glycoprotein (MOG) antibody (ab) related optic neuritis (ON) tend to have characteristics that differ from seronegative ones. The aim of our study was to investigate the clinical characteristics of pediatric anti-MOG ON by comparing anti MOG-ab-seropositive and seronegative patients with ON. in this retrospective Italian multicentre study, participants were identified by chart review of patients evaluated for acquired demyelinating syndromes of the central nervous system (over the period 2009-2019). We selected patients presenting with ON as their first demyelinating event. Inclusion criteria were age < 18 years at symptoms onset; presentation consistent with ON; negativity of anti-aquaporin 4 antibodies (AQP4). Only patients who were tested for MOG-IgG1-ab with a live cell-based assay were included. 22 patients (10 MOG-ab-positive and 12 MOG-ab-negative) were included. Fundus oculi examination at onset showed disc swelling in 9/10 in the MOG-ab-positive cohort and 2/10 in the seronegative group (P = 0.002). Retinal Fiber Nerve Layer (RFNL) thickness measured by Spectral Domain Optical Coherence Tomography (S-OCT) was increased in the 5/5 MOG-ab-positive patients tested and was normal or reduced in the seronegative patients tested (4/4 patients) (P = 0.024). Visual acuity impairment at onset did not differ significantly between the two groups, but the MOG-ab-positive cohort showed better recovery at follow-up both regarding visual acuity (P = 0.025) and expanded disability status scale (EDSS) (P = 0.013). A final diagnosis of MS was frequent among seronegative patients (6/12, 50%), whereas none of the MOG-ab-positive group received a diagnosis of MS (P = 0.015). Clinical relapse frequency was low in both groups: 2/10 MOG-ab-positive and 2/12 seronegative cases relapsed, with a median follow up of 25 months. optic disc swelling and increased RFNL at baseline are strongly associated with MOG-ab positivity. MOG-ab-positive patients with ON showed better recovery compared to the seronegative ones. The relapse rate was low and did not differ among the two groups.

Sections du résumé

BACKGROUND BACKGROUND
recent studies reported that anti myelin oligodendrocyte glycoprotein (MOG) antibody (ab) related optic neuritis (ON) tend to have characteristics that differ from seronegative ones. The aim of our study was to investigate the clinical characteristics of pediatric anti-MOG ON by comparing anti MOG-ab-seropositive and seronegative patients with ON.
METHODS METHODS
in this retrospective Italian multicentre study, participants were identified by chart review of patients evaluated for acquired demyelinating syndromes of the central nervous system (over the period 2009-2019). We selected patients presenting with ON as their first demyelinating event. Inclusion criteria were age < 18 years at symptoms onset; presentation consistent with ON; negativity of anti-aquaporin 4 antibodies (AQP4). Only patients who were tested for MOG-IgG1-ab with a live cell-based assay were included.
RESULTS RESULTS
22 patients (10 MOG-ab-positive and 12 MOG-ab-negative) were included. Fundus oculi examination at onset showed disc swelling in 9/10 in the MOG-ab-positive cohort and 2/10 in the seronegative group (P = 0.002). Retinal Fiber Nerve Layer (RFNL) thickness measured by Spectral Domain Optical Coherence Tomography (S-OCT) was increased in the 5/5 MOG-ab-positive patients tested and was normal or reduced in the seronegative patients tested (4/4 patients) (P = 0.024). Visual acuity impairment at onset did not differ significantly between the two groups, but the MOG-ab-positive cohort showed better recovery at follow-up both regarding visual acuity (P = 0.025) and expanded disability status scale (EDSS) (P = 0.013). A final diagnosis of MS was frequent among seronegative patients (6/12, 50%), whereas none of the MOG-ab-positive group received a diagnosis of MS (P = 0.015). Clinical relapse frequency was low in both groups: 2/10 MOG-ab-positive and 2/12 seronegative cases relapsed, with a median follow up of 25 months.
CONCLUSION CONCLUSIONS
optic disc swelling and increased RFNL at baseline are strongly associated with MOG-ab positivity. MOG-ab-positive patients with ON showed better recovery compared to the seronegative ones. The relapse rate was low and did not differ among the two groups.

Identifiants

DOI: 10.1016/j.msard.2019.101917 PMID: 31896061
pubmed: 31896061
pii: S2211-0348(19)30988-5
doi: 10.1016/j.msard.2019.101917
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

101917

Informations de copyright

Copyright © 2020 Elsevier B.V. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Auteurs

Thea Giacomini (T)

Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Thomas Foiadelli (T)

Pediatric Clinic, IRCCS Policlinico San Matteo Foundation, Pavia, Italy; Department of Clinical-Surgical, Diagnostic and Pediatric Sciences, University of Pavia, Italy.

Pietro Annovazzi (P)

Multiple Sclerosis Center. ASST Valle Olona - Gallarate Hospital, Gallarate (VA), Italy.

Margherita Nosadini (M)

Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy.

Matteo Gastaldi (M)

Neuroimmunology Laboratory, IRCCS Mondino Foundation, Pavia, Italy.

Diego Franciotta (D)

Neuroimmunology Laboratory, IRCCS Mondino Foundation, Pavia, Italy.

Claudio Panarese (C)

CSF Laboratory, Ospedale Policlinico San Martino-IRCCS, Genoa, Italy; Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.

Paolo Capris (P)

Unit of Ophthalmology, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Paola Camicione (P)

Unit of Ophthalmology, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Paola Lanteri (P)

Unit of Neurophisyology, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Elisa De Grandis (E)

Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy; Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.

Giulia Prato (G)

Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Ramona Cordani (R)

Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.

Lino Nobili (L)

Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy; Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.

Giovanni Morana (G)

Unit of Neuroradiology, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Andrea Rossi (A)

Unit of Neuroradiology, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Angela Pistorio (A)

Epidemiology and Biostatistics Unit, IRCCS Istituto Giannina Gaslini, Genova, Italy.

Maria Cellerino (M)

Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.

Antonio Uccelli (A)

Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy; Ospedale Policlinico San Martino-IRCCS, Genoa, Italy.

Stefano Sartori (S)

Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy.

Salvatore Savasta (S)

Pediatric Clinic, IRCCS Policlinico San Matteo Foundation, Pavia, Italy; Department of Clinical-Surgical, Diagnostic and Pediatric Sciences, University of Pavia, Italy.

Maria Margherita Mancardi (MM)

Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy. Electronic address: margheritamancardi@gaslini.org.

Classifications MeSH