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 Sartori; Salvatore Savasta; Maria Margherita Mancardi

doi:10.1016/j.msard.2019.101917

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

Mult Scler Relat Disord. 2020 Apr:39:101917. doi: 10.1016/j.msard.2019.101917. Epub 2019 Dec 24.

Authors

Thea Giacomini¹, Thomas Foiadelli², Pietro Annovazzi³, Margherita Nosadini⁴, Matteo Gastaldi⁵, Diego Franciotta⁵, Claudio Panarese⁶, Paolo Capris⁷, Paola Camicione⁷, Paola Lanteri⁸, Elisa De Grandis⁹, Giulia Prato¹, Ramona Cordani¹⁰, Lino Nobili⁹, Giovanni Morana¹¹, Andrea Rossi¹¹, Angela Pistorio¹², Maria Cellerino¹⁰, Antonio Uccelli¹³, Stefano Sartori⁴, Salvatore Savasta², Maria Margherita Mancardi¹⁴

Affiliations

¹ Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy.
² Pediatric Clinic, IRCCS Policlinico San Matteo Foundation, Pavia, Italy; Department of Clinical-Surgical, Diagnostic and Pediatric Sciences, University of Pavia, Italy.
³ Multiple Sclerosis Center. ASST Valle Olona - Gallarate Hospital, Gallarate (VA), Italy.
⁴ Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy.
⁵ Neuroimmunology Laboratory, IRCCS Mondino Foundation, Pavia, Italy.
⁶ CSF Laboratory, Ospedale Policlinico San Martino-IRCCS, Genoa, Italy; Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.
⁷ Unit of Ophthalmology, IRCCS Istituto Giannina Gaslini, Genova, Italy.
⁸ Unit of Neurophisyology, IRCCS Istituto Giannina Gaslini, Genova, Italy.
⁹ 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.
¹⁰ Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy.
¹¹ Unit of Neuroradiology, IRCCS Istituto Giannina Gaslini, Genova, Italy.
¹² Epidemiology and Biostatistics Unit, IRCCS Istituto Giannina Gaslini, Genova, Italy.
¹³ Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genoa, Italy; Ospedale Policlinico San Martino-IRCCS, Genoa, Italy.
¹⁴ Unit of Child Neuropsychiatry, Clinical and Surgical Neurosciences Department, IRCCS Istituto Giannina Gaslini, Genova, Italy. Electronic address: margheritamancardi@gaslini.org.

PMID: 31896061
DOI: 10.1016/j.msard.2019.101917

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.

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