Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes

Introduction: The relationship between serum neurofilament light chain (sNfL) and myelin oligodendrocyte glycoprotein antibody (MOG-Ab) status has not been yet investigated in children with the acquired demyelinating syndrome (ADS).Objective and Methods: The sNfL levels and MOG-Abs were measured by...

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Autores principales:	Marta Simone, Claudia Palazzo, Mariangela Mastrapasqua, Luca Bollo, Francesco Pompamea, Alessandra Gabellone, Lucia Marzulli, Paola Giordano, Andrea De Giacomo, Antonio Frigeri, Maddalena Ruggieri, Lucia Margari
Formato:	article
Lenguaje:	EN
Publicado:	Frontiers Media S.A. 2021
Materias:	serum neurofilament light chain encephalopathy myelin oligodendrocyte antibody pediatric acute demyelinating disease biomarkers Neurology. Diseases of the nervous system RC346-429
Acceso en línea:	https://doaj.org/article/60b491ac5d75446f95a9a4b62d91e07f
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spelling	oai:doaj.org-article:60b491ac5d75446f95a9a4b62d91e07f2021-11-11T05:02:03ZSerum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes1664-229510.3389/fneur.2021.754518https://doaj.org/article/60b491ac5d75446f95a9a4b62d91e07f2021-11-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fneur.2021.754518/fullhttps://doaj.org/toc/1664-2295Introduction: The relationship between serum neurofilament light chain (sNfL) and myelin oligodendrocyte glycoprotein antibody (MOG-Ab) status has not been yet investigated in children with the acquired demyelinating syndrome (ADS).Objective and Methods: The sNfL levels and MOG-Abs were measured by ultrasensitive single-molecule array and cell-based assay in a cohort of 37 children with ADS and negativity for serum anti-aquaporin 4 (AQP4) antibodies. The sNfL levels were compared in MOG-Ab+/MOG-Ab– and in two subgroups MOG-Ab+ with/without encephalopathy.Results: About 40% ADS resulted in MOG-Ab+. MOG-Ab+ were younger at sampling (median = 9.8; range = 2.17–17.5 vs. 14.7/9–17; p = 0.002) with lower frequency of cerebrospinal fluid oligoclonal bands positivity (27% vs. 70%; p = 0.013) compared to MOG-Ab–. About 53% of MOG-Ab+ presented encephalopathy at onset, 1/22 of MOG-Ab– (p = 0.0006). Higher sNfL levels (p = 0.0001) were found in MOG-Ab+ (median/range = 11.11/6.8–1,129) and MOG-Ab– (median/range = 11.6/4.3–788) compared to age-matched controls (median/range = 2.98/1–4.53), without significant difference. MOG-Ab+ with encephalopathy resulted significantly younger at sampling (median/range: 4.5/2.17–11.17 vs. 14.16/9.8–17.5; p = 0.004), had higher sNfL levels (median/range:75.24/9.1–1,129 vs. 10.22/6.83–50.53; p = 0.04), and showed a trend for higher MOG-Ab titer (0.28/0.04–0.69 vs. 0.05/0.04–0.28; p = 0.1) in comparison to those without encephalopathy.Discussion: We confirmed high sNfL levels in pediatric ADS independently from the MOG-Ab status. Encephalopathy at onset is associated more frequently with MOG Ab+ children with higher sNfL levels and MOG titer. These findings suggest a role of acute demyelination in association with axonal damage in the pathogenesis of encephalopathy in pediatric ADS.Marta SimoneClaudia PalazzoMariangela MastrapasquaLuca BolloFrancesco PompameaAlessandra GabelloneLucia MarzulliPaola GiordanoAndrea De GiacomoAntonio FrigeriMaddalena RuggieriLucia MargariFrontiers Media S.A.articleserum neurofilament light chainencephalopathymyelin oligodendrocyte antibodypediatric acute demyelinating diseasebiomarkersNeurology. Diseases of the nervous systemRC346-429ENFrontiers in Neurology, Vol 12 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	serum neurofilament light chain encephalopathy myelin oligodendrocyte antibody pediatric acute demyelinating disease biomarkers Neurology. Diseases of the nervous system RC346-429
spellingShingle	serum neurofilament light chain encephalopathy myelin oligodendrocyte antibody pediatric acute demyelinating disease biomarkers Neurology. Diseases of the nervous system RC346-429 Marta Simone Claudia Palazzo Mariangela Mastrapasqua Luca Bollo Francesco Pompamea Alessandra Gabellone Lucia Marzulli Paola Giordano Andrea De Giacomo Antonio Frigeri Maddalena Ruggieri Lucia Margari Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes
description	Introduction: The relationship between serum neurofilament light chain (sNfL) and myelin oligodendrocyte glycoprotein antibody (MOG-Ab) status has not been yet investigated in children with the acquired demyelinating syndrome (ADS).Objective and Methods: The sNfL levels and MOG-Abs were measured by ultrasensitive single-molecule array and cell-based assay in a cohort of 37 children with ADS and negativity for serum anti-aquaporin 4 (AQP4) antibodies. The sNfL levels were compared in MOG-Ab+/MOG-Ab– and in two subgroups MOG-Ab+ with/without encephalopathy.Results: About 40% ADS resulted in MOG-Ab+. MOG-Ab+ were younger at sampling (median = 9.8; range = 2.17–17.5 vs. 14.7/9–17; p = 0.002) with lower frequency of cerebrospinal fluid oligoclonal bands positivity (27% vs. 70%; p = 0.013) compared to MOG-Ab–. About 53% of MOG-Ab+ presented encephalopathy at onset, 1/22 of MOG-Ab– (p = 0.0006). Higher sNfL levels (p = 0.0001) were found in MOG-Ab+ (median/range = 11.11/6.8–1,129) and MOG-Ab– (median/range = 11.6/4.3–788) compared to age-matched controls (median/range = 2.98/1–4.53), without significant difference. MOG-Ab+ with encephalopathy resulted significantly younger at sampling (median/range: 4.5/2.17–11.17 vs. 14.16/9.8–17.5; p = 0.004), had higher sNfL levels (median/range:75.24/9.1–1,129 vs. 10.22/6.83–50.53; p = 0.04), and showed a trend for higher MOG-Ab titer (0.28/0.04–0.69 vs. 0.05/0.04–0.28; p = 0.1) in comparison to those without encephalopathy.Discussion: We confirmed high sNfL levels in pediatric ADS independently from the MOG-Ab status. Encephalopathy at onset is associated more frequently with MOG Ab+ children with higher sNfL levels and MOG titer. These findings suggest a role of acute demyelination in association with axonal damage in the pathogenesis of encephalopathy in pediatric ADS.
format	article
author	Marta Simone Claudia Palazzo Mariangela Mastrapasqua Luca Bollo Francesco Pompamea Alessandra Gabellone Lucia Marzulli Paola Giordano Andrea De Giacomo Antonio Frigeri Maddalena Ruggieri Lucia Margari
author_facet	Marta Simone Claudia Palazzo Mariangela Mastrapasqua Luca Bollo Francesco Pompamea Alessandra Gabellone Lucia Marzulli Paola Giordano Andrea De Giacomo Antonio Frigeri Maddalena Ruggieri Lucia Margari
author_sort	Marta Simone
title	Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes
title_short	Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes
title_full	Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes
title_fullStr	Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes
title_full_unstemmed	Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes
title_sort	serum neurofilament light chain levels and myelin oligodendrocyte glycoprotein antibodies in pediatric acquired demyelinating syndromes
publisher	Frontiers Media S.A.
publishDate	2021
url	https://doaj.org/article/60b491ac5d75446f95a9a4b62d91e07f
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Serum Neurofilament Light Chain Levels and Myelin Oligodendrocyte Glycoprotein Antibodies in Pediatric Acquired Demyelinating Syndromes

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