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Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease

BACKGROUND AND OBJECTIVES: Elevated intracranial pressure (ICP) in myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease (MOGAD) has been largely unexplored. The objectives of this study were to determine the frequency of increased ICP in MOGAD and its association with disease course...

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Autores principales: Nguyen, Linda, Miles, Darryl K., Harder, Lana, Singh, Sumit, Whittemore, Brett A., Greenberg, Benjamin M., Wang, Cynthia X.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10621892/
https://www.ncbi.nlm.nih.gov/pubmed/37918972
http://dx.doi.org/10.1212/NXI.0000000000200174
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author Nguyen, Linda
Miles, Darryl K.
Harder, Lana
Singh, Sumit
Whittemore, Brett A.
Greenberg, Benjamin M.
Wang, Cynthia X.
author_facet Nguyen, Linda
Miles, Darryl K.
Harder, Lana
Singh, Sumit
Whittemore, Brett A.
Greenberg, Benjamin M.
Wang, Cynthia X.
author_sort Nguyen, Linda
collection PubMed
description BACKGROUND AND OBJECTIVES: Elevated intracranial pressure (ICP) in myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease (MOGAD) has been largely unexplored. The objectives of this study were to determine the frequency of increased ICP in MOGAD and its association with disease course and outcomes and to highlight cases requiring medical and/or surgical management of increased ICP. METHODS: In this retrospective, single-center cohort study, we examined the clinical and paraclinical data from the initial presentation and follow-up data of children diagnosed with MOGAD. In those with opening pressure (OP) measurements, univariate analyses were used to evaluate factors associated with increased ICP, which was defined as OP > 28 cm H(2)O. We also present a case series of patients with or without OP measurement who required medical and/or surgical management of increased ICP. RESULTS: Of 86 children with MOGAD, 43 (50.0%) had an OP recorded and 7 (8.1%) required ICP management. In those with OP recorded, the median (interquartile range) OP for the different MOGAD phenotypes were: 30.0 (22.8–41.6) (acute disseminated encephalomyelitis, ADEM), 20.5 (16.1–23.6) (optic neuritis), 17.0 (17.0–22.5) (myelitis), and 19.5 (16.5–29.3) (other) cm H(2)0. Overall, 20.9% had increased ICP based on an OP > 28 cm H(2)O, of whom 77.8% presented with ADEM. In a subgroup analysis of those presenting with ADEM, those with an elevated ICP had longer hospital stay (p = 0.007) and neurologic disability (defined as modified Rankin Scale >1) (p = 0.049). In those with or without OP recorded, 7 (6 with ADEM, one with cerebral cortical encephalitis) required ICP-directed therapies. Findings on brain MRI in these 7 children revealed extensive disease burden with bilateral cerebral involvement and evidence of restricted diffusion. While neuropsychological data in this small subset revealed significant variability, all sustained identifiable deficits after discharge, including attention-deficit hyperactivity disorders and language and learning disorders. DISCUSSION: In pediatric MOGAD, increased OP and ADEM at initial presentation were associated with longer hospital stays and greater long-term morbidity. Although invasive ICP monitoring has not been specifically advocated in the management of MOGAD, it is important to recognize signs and symptoms of increased ICP in these patients and consider ICP monitoring and management strategies based on clinical and radiologic findings, especially in those presenting with ADEM and with OP > 28 cm H(2)O.
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spelling pubmed-106218922023-11-03 Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease Nguyen, Linda Miles, Darryl K. Harder, Lana Singh, Sumit Whittemore, Brett A. Greenberg, Benjamin M. Wang, Cynthia X. Neurol Neuroimmunol Neuroinflamm Research Article BACKGROUND AND OBJECTIVES: Elevated intracranial pressure (ICP) in myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease (MOGAD) has been largely unexplored. The objectives of this study were to determine the frequency of increased ICP in MOGAD and its association with disease course and outcomes and to highlight cases requiring medical and/or surgical management of increased ICP. METHODS: In this retrospective, single-center cohort study, we examined the clinical and paraclinical data from the initial presentation and follow-up data of children diagnosed with MOGAD. In those with opening pressure (OP) measurements, univariate analyses were used to evaluate factors associated with increased ICP, which was defined as OP > 28 cm H(2)O. We also present a case series of patients with or without OP measurement who required medical and/or surgical management of increased ICP. RESULTS: Of 86 children with MOGAD, 43 (50.0%) had an OP recorded and 7 (8.1%) required ICP management. In those with OP recorded, the median (interquartile range) OP for the different MOGAD phenotypes were: 30.0 (22.8–41.6) (acute disseminated encephalomyelitis, ADEM), 20.5 (16.1–23.6) (optic neuritis), 17.0 (17.0–22.5) (myelitis), and 19.5 (16.5–29.3) (other) cm H(2)0. Overall, 20.9% had increased ICP based on an OP > 28 cm H(2)O, of whom 77.8% presented with ADEM. In a subgroup analysis of those presenting with ADEM, those with an elevated ICP had longer hospital stay (p = 0.007) and neurologic disability (defined as modified Rankin Scale >1) (p = 0.049). In those with or without OP recorded, 7 (6 with ADEM, one with cerebral cortical encephalitis) required ICP-directed therapies. Findings on brain MRI in these 7 children revealed extensive disease burden with bilateral cerebral involvement and evidence of restricted diffusion. While neuropsychological data in this small subset revealed significant variability, all sustained identifiable deficits after discharge, including attention-deficit hyperactivity disorders and language and learning disorders. DISCUSSION: In pediatric MOGAD, increased OP and ADEM at initial presentation were associated with longer hospital stays and greater long-term morbidity. Although invasive ICP monitoring has not been specifically advocated in the management of MOGAD, it is important to recognize signs and symptoms of increased ICP in these patients and consider ICP monitoring and management strategies based on clinical and radiologic findings, especially in those presenting with ADEM and with OP > 28 cm H(2)O. Lippincott Williams & Wilkins 2023-10-31 /pmc/articles/PMC10621892/ /pubmed/37918972 http://dx.doi.org/10.1212/NXI.0000000000200174 Text en Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND) (https://creativecommons.org/licenses/by-nc-nd/4.0/) , which permits downloading and sharing the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.
spellingShingle Research Article
Nguyen, Linda
Miles, Darryl K.
Harder, Lana
Singh, Sumit
Whittemore, Brett A.
Greenberg, Benjamin M.
Wang, Cynthia X.
Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease
title Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease
title_full Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease
title_fullStr Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease
title_full_unstemmed Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease
title_short Increased Intracranial Pressure in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease
title_sort increased intracranial pressure in pediatric myelin oligodendrocyte glycoprotein antibody–associated disease
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10621892/
https://www.ncbi.nlm.nih.gov/pubmed/37918972
http://dx.doi.org/10.1212/NXI.0000000000200174
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