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Title:
Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well? | Journal of Neuroinflammation
Description:
Background To investigate age-related severity, patterns of retinal structural damage, and functional visual recovery in pediatric and adult cohorts of myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) optic neuritis (ON). Methods All MOGAD patients from the 5 participating centers were included. Patients with initial manifestation <18 years were included in the pediatric (MOGADped) cohort and patients with ≥18 years in the adult (MOGADadult) cohort. For patients with MOGAD ON, examinations at least ≥6 months after ON onset were included in the analyses. Using spectral domain optical coherence tomography (SD-OCT), we acquired peripapillary retinal nerve fiber layer thickness (pRNFL) and volumes of combined ganglion cell and inner plexiform layer (GCIPL). High- and 2.5% low-contrast visual acuity (HCVA, LCVA) and visual-evoked potentials (VEP) were obtained. Results Twenty MOGADped (10.3±3.7 years, 30 MOGAD ON eyes) and 39 MOGADadult (34.9±11.6 years, 42 MOGAD ON eyes) patients were included. The average number of ON episodes per ON eye was similar in both groups (1.8±1.3 and 2.0±1.7). In both pediatric and adult MOGAD, ON led to pronounced neuroaxonal retinal atrophy (pRNFL: 63.1±18.7 and 64.3±22.9 μm; GCIPL: 0.42±0.09 and 0.44±0.13 mm3, respectively) and moderate delay of the VEP latencies (117.9±10.7 and 118.0±14.5 ms). In contrast, visual acuity was substantially better in children (HCVA: 51.4±9.3 vs. 35.0±20.6 raw letters, p=0.001; LCVA: 22.8±14.6 vs. 13.5±16.4, p=0.028). Complete visual recovery (HCVA-logMAR 0.0) occurred in 73.3% of MOGADped and 31% MOGADadults ON eyes, while 3.3% and 31% demonstrated moderate to severe (logMAR > 0.5) visual impairment. Independent of retinal atrophy, age at ON onset significantly correlated with visual outcome. Conclusion Pediatric MOGAD ON showed better visual recovery than adult MOGAD ON despite profound and almost identical neuroaxonal retinal atrophy. Age-related cortical neuroplasticity may account for the substantial discrepancy between structural changes and functional outcomes.
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Keywords {🔍}
patients, visual, article, pubmed, mogad, google, scholar, eyes, retinal, study, data, clinical, pediatric, optic, prnfl, children, atrophy, age, recovery, mogadped, adult, hcva, vep, neuritis, mogadadult, outcome, germany, volume, myelin, oligodendrocyte, cohort, lcva, neuroaxonal, central, cas, neurol, years, macular, hospital, number, glycoprotein, nerve, layer, mog, adults, oct, gcipl, compared, university, analysis,
Topics {✒️}
myelin-oligodendrocyte-glycoprotein antibody-seropositive patients myelin oligodendrocyte glycoprotein-specific myelin oligodendrocyte glycoprotein myelin-oligodendrocyte-glycoprotein optical coherence tomography clinical profile central nervous system aqp-4 igg-seropositive nmosd aqp4-igg seropositive nmosd view lcva low-contrast visusal acuity retro-illuminated light box ludwig-maximilians universität münchen guthy-jackson charitable foundation blood-brain barrier properties post-geniculate visual pathways bennett marianne-strauß-klinik grant numbers 01zz1603[a aqp4-abs-positive nmosd article download pdf pmb paillo-macular-bundle 20 mog-igg-positive patients 39 mog-igg-positive patients aquaporin-4-igg-positive nmosd established cell-based assays visual-evoked potentials active age-dependent neuroplasticity 39 mog-igg-positive adults 20 mog-igg-positive children age-dependent cortical neuroplasticity patient inter-eye correlation markus reindl adult retinal oct scans optic nerve lesions investigate age-related severity severe optic neuritis childhood optic neuritis recurrent optic neuritis joachim havla pediatric optic neuritis ana felipe-rucián age-related cortical neuroplasticity original author neuroimmunological diseases bilateral optic neuritis optic neuritis independent lotz-havla department article havla privacy choices/manage cookies relapsing mog antibody
Questions {❓}
- Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well?
- Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well?
- Is a preserved functional reserve a mechanism limiting clinical impairment in pediatric MS patients?
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headline:Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well?
description:To investigate age-related severity, patterns of retinal structural damage, and functional visual recovery in pediatric and adult cohorts of myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) optic neuritis (ON). All MOGAD patients from the 5 participating centers were included. Patients with initial manifestation <18 years were included in the pediatric (MOGADped) cohort and patients with ≥18 years in the adult (MOGADadult) cohort. For patients with MOGAD ON, examinations at least ≥6 months after ON onset were included in the analyses. Using spectral domain optical coherence tomography (SD-OCT), we acquired peripapillary retinal nerve fiber layer thickness (pRNFL) and volumes of combined ganglion cell and inner plexiform layer (GCIPL). High- and 2.5% low-contrast visual acuity (HCVA, LCVA) and visual-evoked potentials (VEP) were obtained. Twenty MOGADped (10.3±3.7 years, 30 MOGAD ON eyes) and 39 MOGADadult (34.9±11.6 years, 42 MOGAD ON eyes) patients were included. The average number of ON episodes per ON eye was similar in both groups (1.8±1.3 and 2.0±1.7). In both pediatric and adult MOGAD, ON led to pronounced neuroaxonal retinal atrophy (pRNFL: 63.1±18.7 and 64.3±22.9 μm; GCIPL: 0.42±0.09 and 0.44±0.13 mm3, respectively) and moderate delay of the VEP latencies (117.9±10.7 and 118.0±14.5 ms). In contrast, visual acuity was substantially better in children (HCVA: 51.4±9.3 vs. 35.0±20.6 raw letters, p=0.001; LCVA: 22.8±14.6 vs. 13.5±16.4, p=0.028). Complete visual recovery (HCVA-logMAR 0.0) occurred in 73.3% of MOGADped and 31% MOGADadults ON eyes, while 3.3% and 31% demonstrated moderate to severe (logMAR > 0.5) visual impairment. Independent of retinal atrophy, age at ON onset significantly correlated with visual outcome. Pediatric MOGAD ON showed better visual recovery than adult MOGAD ON despite profound and almost identical neuroaxonal retinal atrophy. Age-related cortical neuroplasticity may account for the substantial discrepancy between structural changes and functional outcomes.
datePublished:2021-05-29T00:00:00Z
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Optical coherence tomography
Optic neuritis
Myelin oligodendrocyte glycoprotein IgG
MOGAD
Neurosciences
Neurology
Neurobiology
Immunology
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headline:Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well?
description:To investigate age-related severity, patterns of retinal structural damage, and functional visual recovery in pediatric and adult cohorts of myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) optic neuritis (ON). All MOGAD patients from the 5 participating centers were included. Patients with initial manifestation <18 years were included in the pediatric (MOGADped) cohort and patients with ≥18 years in the adult (MOGADadult) cohort. For patients with MOGAD ON, examinations at least ≥6 months after ON onset were included in the analyses. Using spectral domain optical coherence tomography (SD-OCT), we acquired peripapillary retinal nerve fiber layer thickness (pRNFL) and volumes of combined ganglion cell and inner plexiform layer (GCIPL). High- and 2.5% low-contrast visual acuity (HCVA, LCVA) and visual-evoked potentials (VEP) were obtained. Twenty MOGADped (10.3±3.7 years, 30 MOGAD ON eyes) and 39 MOGADadult (34.9±11.6 years, 42 MOGAD ON eyes) patients were included. The average number of ON episodes per ON eye was similar in both groups (1.8±1.3 and 2.0±1.7). In both pediatric and adult MOGAD, ON led to pronounced neuroaxonal retinal atrophy (pRNFL: 63.1±18.7 and 64.3±22.9 μm; GCIPL: 0.42±0.09 and 0.44±0.13 mm3, respectively) and moderate delay of the VEP latencies (117.9±10.7 and 118.0±14.5 ms). In contrast, visual acuity was substantially better in children (HCVA: 51.4±9.3 vs. 35.0±20.6 raw letters, p=0.001; LCVA: 22.8±14.6 vs. 13.5±16.4, p=0.028). Complete visual recovery (HCVA-logMAR 0.0) occurred in 73.3% of MOGADped and 31% MOGADadults ON eyes, while 3.3% and 31% demonstrated moderate to severe (logMAR > 0.5) visual impairment. Independent of retinal atrophy, age at ON onset significantly correlated with visual outcome. Pediatric MOGAD ON showed better visual recovery than adult MOGAD ON despite profound and almost identical neuroaxonal retinal atrophy. Age-related cortical neuroplasticity may account for the substantial discrepancy between structural changes and functional outcomes.
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Optical coherence tomography
Optic neuritis
Myelin oligodendrocyte glycoprotein IgG
MOGAD
Neurosciences
Neurology
Neurobiology
Immunology
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address:
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type:PostalAddress
type:Organization
email:[email protected]
name:Thivya Pakeerathan
affiliation:
name:Ruhr-University Bochum
address:
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Carolin Schwake
affiliation:
name:Ruhr-University Bochum
address:
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Jeffrey L. Bennett
affiliation:
name:University of Colorado Anschutz Medical Campus
address:
name:Departments of Neurology and Ophthalmology, Programs in Neuroscience and Immunology, University of Colorado Anschutz Medical Campus, Denver, USA
type:PostalAddress
type:Organization
name:Ingo Kleiter
affiliation:
name:Ruhr-University Bochum
address:
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Behandlungszentrum Kempfenhausen für Multiple Sklerose Kranke
address:
name:Marianne-Strauß-Klinik, Behandlungszentrum Kempfenhausen für Multiple Sklerose Kranke, Berg, Germany
type:PostalAddress
type:Organization
name:Ana Felipe-Rucián
affiliation:
name:Universitat Autònoma de Barcelona
address:
name:Department of Pediatric Neurology, Vall d’Hebron Hospital, Universitat Autònoma de Barcelona, Barcelona, Spain
type:PostalAddress
type:Organization
name:Stephanie C. Joachim
affiliation:
name:Ruhr-University Bochum
address:
name:Experimental Eye Research Institute, University Eye Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Amelie S. Lotz-Havla
affiliation:
name:Ludwig-Maximilians Universität München
address:
name:Dr. von Hauner Children’s Hospital, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany
type:PostalAddress
type:Organization
name:Tania Kümpfel
affiliation:
name:Ludwig-Maximilians Universität München
address:
name:Institute of Clinical Neuroimmunology, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany
type:PostalAddress
type:Organization
name:Markus Krumbholz
affiliation:
name:University Hospital of Tübingen
address:
name:Department of Neurology & Stroke and Hertie Institute for Clinical Brain Research, University Hospital of Tübingen, Tübingen, Germany
type:PostalAddress
type:Organization
name:Eva M. Wendel
affiliation:
name:Olgaspital Stuttgart
address:
name:Department of Pediatric Neurology, Olgaspital Stuttgart, Stuttgart, Germany
type:PostalAddress
type:Organization
name:Markus Reindl
affiliation:
name:Medical University of Innsbruck
address:
name:Clinical Department of Neurology, Medical University of Innsbruck, Innsbruck, Austria
type:PostalAddress
type:Organization
name:Charlotte Thiels
affiliation:
name:Ruhr-University
address:
name:Department of Neuropaediatrics and Social Pediatrics, University Hospital of Pediatrics and Adolescent Medicine, Ruhr-University, Bochum, Germany
type:PostalAddress
type:Organization
name:Thomas Lücke
affiliation:
name:Ruhr-University
address:
name:Department of Neuropaediatrics and Social Pediatrics, University Hospital of Pediatrics and Adolescent Medicine, Ruhr-University, Bochum, Germany
type:PostalAddress
type:Organization
name:Kerstin Hellwig
affiliation:
name:Ruhr-University Bochum
address:
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Ralf Gold
affiliation:
name:Ruhr-University Bochum
address:
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Kevin Rostasy
affiliation:
name:University Witten/Herdecke
address:
name:Department of Pediatric Neurology, Children’s Hospital Datteln, University Witten/Herdecke, Witten, Germany
type:PostalAddress
type:Organization
name:Ilya Ayzenberg
affiliation:
name:Ruhr-University Bochum
address:
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
type:PostalAddress
type:Organization
name:Sechenov First Moscow State Medical University
address:
name:Department of Neurology, Sechenov First Moscow State Medical University, Moscow, Russia
type:PostalAddress
type:Organization
email:[email protected]
PostalAddress:
name:Institute of Clinical Neuroimmunology, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany
name:Data Integration for Future Medicine (DIFUTURE) Consortium, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
name:Departments of Neurology and Ophthalmology, Programs in Neuroscience and Immunology, University of Colorado Anschutz Medical Campus, Denver, USA
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
name:Marianne-Strauß-Klinik, Behandlungszentrum Kempfenhausen für Multiple Sklerose Kranke, Berg, Germany
name:Department of Pediatric Neurology, Vall d’Hebron Hospital, Universitat Autònoma de Barcelona, Barcelona, Spain
name:Experimental Eye Research Institute, University Eye Hospital, Ruhr-University Bochum, Bochum, Germany
name:Dr. von Hauner Children’s Hospital, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany
name:Institute of Clinical Neuroimmunology, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany
name:Department of Neurology & Stroke and Hertie Institute for Clinical Brain Research, University Hospital of Tübingen, Tübingen, Germany
name:Department of Pediatric Neurology, Olgaspital Stuttgart, Stuttgart, Germany
name:Clinical Department of Neurology, Medical University of Innsbruck, Innsbruck, Austria
name:Department of Neuropaediatrics and Social Pediatrics, University Hospital of Pediatrics and Adolescent Medicine, Ruhr-University, Bochum, Germany
name:Department of Neuropaediatrics and Social Pediatrics, University Hospital of Pediatrics and Adolescent Medicine, Ruhr-University, Bochum, Germany
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
name:Department of Pediatric Neurology, Children’s Hospital Datteln, University Witten/Herdecke, Witten, Germany
name:Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany
name:Department of Neurology, Sechenov First Moscow State Medical University, Moscow, Russia
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