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

Thea Giacomini, Thomas Foiadelli, Pietro Annovazzi, Margherita Nosadini, Matteo Gastaldi, Diego Franciotta, Claudio Panarese, Paolo Capris, Paola Camicione, Paola Lanteri, Elisa De Grandis, Giulia Prato, Ramona Cordani, Lino Nobili, Giovanni Morana, Andrea Rossi, Angela Pistorio, Maria Cellerino, Antonio Uccelli, Stefano SartoriSalvatore Savasta, Maria Margherita Mancardi

Research output: Contribution to journalArticle

Abstract

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: 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: 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: 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.

Original languageEnglish
Pages (from-to)101917
JournalMultiple Sclerosis and Related Disorders
Volume39
DOIs
Publication statusE-pub ahead of print - Dec 24 2019

Fingerprint

Myelin-Oligodendrocyte Glycoprotein
Optic Neuritis
Pediatrics
Antibodies
Nerve Fibers
Visual Acuity
Fundus Oculi
Aquaporin 4
Recurrence
Vision Disorders
Optic Disk
Optical Coherence Tomography

Cite this

Pediatric optic neuritis and anti MOG antibodies : a cohort of Italian patients. / Giacomini, Thea; Foiadelli, Thomas; Annovazzi, Pietro; Nosadini, Margherita; Gastaldi, Matteo; Franciotta, Diego; Panarese, Claudio; Capris, Paolo; Camicione, Paola; Lanteri, Paola; De Grandis, Elisa; Prato, Giulia; Cordani, Ramona; Nobili, Lino; Morana, Giovanni; Rossi, Andrea; Pistorio, Angela; Cellerino, Maria; Uccelli, Antonio; Sartori, Stefano; Savasta, Salvatore; Mancardi, Maria Margherita.

In: Multiple Sclerosis and Related Disorders, Vol. 39, 24.12.2019, p. 101917.

Research output: Contribution to journalArticle

Giacomini, Thea ; Foiadelli, Thomas ; Annovazzi, Pietro ; Nosadini, Margherita ; Gastaldi, Matteo ; Franciotta, Diego ; Panarese, Claudio ; Capris, Paolo ; Camicione, Paola ; Lanteri, Paola ; De Grandis, Elisa ; Prato, Giulia ; Cordani, Ramona ; Nobili, Lino ; Morana, Giovanni ; Rossi, Andrea ; Pistorio, Angela ; Cellerino, Maria ; Uccelli, Antonio ; Sartori, Stefano ; Savasta, Salvatore ; Mancardi, Maria Margherita. / Pediatric optic neuritis and anti MOG antibodies : a cohort of Italian patients. In: Multiple Sclerosis and Related Disorders. 2019 ; Vol. 39. pp. 101917.
@article{d177786ebcb14053bcfd721828a7d392,
title = "Pediatric optic neuritis and anti MOG antibodies: a cohort of Italian patients",
abstract = "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: 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: 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: 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.",
author = "Thea Giacomini and Thomas Foiadelli and Pietro Annovazzi and Margherita Nosadini and Matteo Gastaldi and Diego Franciotta and Claudio Panarese and Paolo Capris and Paola Camicione and Paola Lanteri and {De Grandis}, Elisa and Giulia Prato and Ramona Cordani and Lino Nobili and Giovanni Morana and Andrea Rossi and Angela Pistorio and Maria Cellerino and Antonio Uccelli and Stefano Sartori and Salvatore Savasta and Mancardi, {Maria Margherita}",
note = "Copyright {\circledC} 2019. Published by Elsevier B.V.",
year = "2019",
month = "12",
day = "24",
doi = "10.1016/j.msard.2019.101917",
language = "English",
volume = "39",
pages = "101917",
journal = "Multiple Sclerosis and Related Disorders",
issn = "2211-0348",
publisher = "Elsevier",

}

TY - JOUR

T1 - Pediatric optic neuritis and anti MOG antibodies

T2 - a cohort of Italian patients

AU - Giacomini, Thea

AU - Foiadelli, Thomas

AU - Annovazzi, Pietro

AU - Nosadini, Margherita

AU - Gastaldi, Matteo

AU - Franciotta, Diego

AU - Panarese, Claudio

AU - Capris, Paolo

AU - Camicione, Paola

AU - Lanteri, Paola

AU - De Grandis, Elisa

AU - Prato, Giulia

AU - Cordani, Ramona

AU - Nobili, Lino

AU - Morana, Giovanni

AU - Rossi, Andrea

AU - Pistorio, Angela

AU - Cellerino, Maria

AU - Uccelli, Antonio

AU - Sartori, Stefano

AU - Savasta, Salvatore

AU - Mancardi, Maria Margherita

N1 - Copyright © 2019. Published by Elsevier B.V.

PY - 2019/12/24

Y1 - 2019/12/24

N2 - 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: 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: 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: 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.

AB - 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: 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: 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: 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.

U2 - 10.1016/j.msard.2019.101917

DO - 10.1016/j.msard.2019.101917

M3 - Article

C2 - 31896061

VL - 39

SP - 101917

JO - Multiple Sclerosis and Related Disorders

JF - Multiple Sclerosis and Related Disorders

SN - 2211-0348

ER -