Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well?

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Titel: Age-dependent favorable visual recovery despite significant retinal atrophy in pediatric MOGAD: how much retina do you really need to see well?
Autoren: Havla, Joachim, Pakeerathan, Thivya, Schwake, Carolin, Bennett, Jeffrey L., Kleiter, Ingo, Felipe-Rucián, Ana, Joachim, Stephanie C., Lotz-Havla, Amelie S., Kümpfel, Tania, Krumbholz, Markus, Wendel, Eva M., Reindl, Markus, Thiels, Charlotte, Lücke, Thomas, Hellwig, Kerstin, Gold, Ralf, Rostasy, Kevin, Ayzenberg, Ilya, Universitat Autònoma de Barcelona
Weitere Verfasser: Institut Català de la Salut, [Havla J] Institute of Clinical Neuroimmunology, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany. Data Integration for Future Medicine (DIFUTURE) Consortium, LMU Hospital, Ludwig-Maximilians Universität München, Munich, Germany. [Pakeerathan T, Schwake C] Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany. [Bennett JL] Departments of Neurology and Ophthalmology, Programs in Neuroscience and Immunology, University of Colorado Anschutz Medical Campus, Denver, USA. [Kleiter I] Department of Neurology, St. Josef-Hospital, Ruhr-University Bochum, Bochum, Germany. Marianne-Strauß-Klinik, Behandlungszentrum Kempfenhausen für Multiple Sklerose Kranke, Berg, Germany. [Felipe-Rucián A] Servei de Neurologia Pediàtrica, Vall d’Hebron Hospital Universitari, Barcelona, Spain. Universitat Autònoma de Barcelona, Bellaterra, Spain, Vall d'Hebron Barcelona Hospital Campus
Quelle: J Neuroinflammation
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Universitat Autònoma de Barcelona
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Journal of Neuroinflammation, Vol 18, Iss 1, Pp 1-10 (2021)
Verlagsinformationen: Springer Science and Business Media LLC, 2021.
Publikationsjahr: 2021
Schlagwörter: Adult, Male, Optic Neuritis, DISEASES::Nervous System Diseases::Neurologic Manifestations::Sensation Disorders::Vision Disorders, Adolescent, Vision Disorders/immunology [MeSH], Age Factors [MeSH], Optic Neuritis/immunology [MeSH], Retinal Degeneration/physiopathology [MeSH], Tomography, Optical Coherence [MeSH], Autoimmune Diseases of the Nervous System/diagnostic imaging [MeSH], Optic neuritis, Cohort Studies [MeSH], Optical coherence tomography, Male [MeSH], Optic Neuritis/complications [MeSH], Retina/diagnostic imaging [MeSH], Myelin-Oligodendrocyte Glycoprotein/immunology [MeSH], MOGAD, Child [MeSH], Visual Acuity/immunology [MeSH], Autoimmune Diseases of the Nervous System/complications [MeSH], Retina/immunology [MeSH], Adolescent [MeSH], Female [MeSH], Retinal Degeneration/immunology [MeSH], Retina/physiopathology [MeSH], Recovery of Function [MeSH], Adult [MeSH], Humans [MeSH], Atrophy/immunology [MeSH], Middle Aged [MeSH], Autoimmune Diseases of the Nervous System/classification [MeSH], Myelin oligodendrocyte glycoprotein IgG, Evoked Potentials, Visual [MeSH], Research, Optic Neuritis/physiopathology [MeSH], Vision Disorders/physiopathology [MeSH], Child, Preschool [MeSH], NAMED GROUPS::Persons::Age Groups::Child, Neurologia pediàtrica, Vision Disorders, Trastorns de la visió, DENOMINACIONES DE GRUPOS::personas::Grupos de Edad::niño, Retina, Cohort Studies, 03 medical and health sciences, Autoimmune Diseases of the Nervous System, 0302 clinical medicine, ENFERMEDADES::enfermedades del sistema nervioso::manifestaciones neurológicas::trastornos sensoriales::trastornos de la visión, Humans, RC346-429, Child, 10. No inequality, Other subheadings::Other subheadings::Other subheadings::/immunology, TÉCNICAS Y EQUIPOS ANALÍTICOS, DIAGNÓSTICOS Y TERAPÉUTICOS::técnicas de investigación::imágenes ópticas::tomografía óptica::tomografía de coherencia óptica, 2. Zero hunger, ANALYTICAL, DIAGNOSTIC AND THERAPEUTIC TECHNIQUES, AND EQUIPMENT::Investigative Techniques::Optical Imaging::Tomography, Optical::Tomography, Optical Coherence, Retina - Malalties - Imatgeria, Retinal Degeneration, Age Factors, Recovery of Function, Middle Aged, 16. Peace & justice, 3. Good health, Otros calificadores::Otros calificadores::Otros calificadores::/inmunología, Child, Preschool, Evoked Potentials, Visual, Female, Myelin-Oligodendrocyte Glycoprotein, Neurology. Diseases of the nervous system, Atrophy, Tomography, Optical Coherence
Beschreibung: 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 ped) 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.
Publikationsart: Article
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Dateibeschreibung: application/pdf
Sprache: English
ISSN: 1742-2094
DOI: 10.1186/s12974-021-02160-9
Zugangs-URL: https://jneuroinflammation.biomedcentral.com/track/pdf/10.1186/s12974-021-02160-9
https://pubmed.ncbi.nlm.nih.gov/34051804
https://ddd.uab.cat/record/255609
https://hdl.handle.net/11351/6842
https://doaj.org/article/e6273de0a81f4441a89f3e0d94a0bee0
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8164737
https://epub.ub.uni-muenchen.de/76549/
https://link.springer.com/content/pdf/10.1186/s12974-021-02160-9.pdf
https://link.springer.com/article/10.1186/s12974-021-02160-9
https://europepmc.org/article/MED/34051804
https://jneuroinflammation.biomedcentral.com/articles/10.1186/s12974-021-02160-9
https://repository.publisso.de/resource/frl:6465778
https://epub.ub.uni-muenchen.de/76549/
Rights: CC BY
Dokumentencode: edsair.doi.dedup.....d7dccc34e4a41d663b5c1d1d364e50d9
Datenbank: OpenAIRE
Beschreibung
Abstract: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 ped) 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.
ISSN:17422094
DOI:10.1186/s12974-021-02160-9