Associations of paediatric demyelinating and encephalitic syndromes with myelin oligodendrocyte glycoprotein antibodies: a multicentre observational study

Armangué, Thaís; Olivé-Cirera, Gemma; Martínez‐Hernández, Eugenia; Sepúlveda, María; Ruiz‐García, Raquel; Muñoz-Batista, Marta; Ariño, Helena; González-Álvarez, Verónica; Felipe‐Rucián, Ana; Martínez-González, Maria Jesús; Cantarín-Extremera, Verónica; Miranda-Herrero, María Concepción; Galindo, L. Monge; Tomás‐Vila, Miguel; Miravet, Elena; Málaga, Ignacio; Arrambide, Georgina; Auger, Cristina; Tintoré, Mar; Montalbán, Xavier; Vanderver, Adeline; Graus, Francesc; Sáiz, Albert; Dalmau, Josep; Alcantud, Alberto; Aguilera-Albesa, Sergio; Demanuel, Diana Alvarez; Molinero, Mireia Alvarez; Fariña, Lourdes Aquino; Arrabal, Luisa; Arriola-Pereda, Gema; Aznar-Laín, Gemma; Benavides-Medina, Maite; Bermejo, Teresa de Jesús; Blanco-Lago, Raquel; Caballero, Eva; Calvo, Rocío; Salas, Ana Camacho; Conejo-Moreno, David; Delgadillo-Chilavert, Verónica; Elosegi-Castellanos, Amagoia; Canto, Vanesa Esteban; Fernández-Ramos, J.A.; Garcia-Puig, Montserrat; García-Ribes, Ainhoa; Gómez-Martín, Hilario; Gonzalez-Barrios, Desire; González‐Gutiérrez‐Solana, Luis; Jimena-Garcia, Sara; Jiménez-Legido, María; Juliá‐Palacios, Natalia; López‐Laso, Eduardo; Martí-Carrera, Itxaso; González, Marta Martínez; Martín-Viota, Lucía; Mattozi, Simone; Maqueda-Castellote, Elena; Mendibe, Maria D M; Mora-Ramírez, Maria D; Muñoz-Cabello, Beatriz; Navarro‐Morón, Juan; Nunes-Cabrera, Tania; Orellana, Gabriela; Pujol-Soler, Berta; Querol, Luís; Ramı́rez, Alfredo; Rodriguez-Lucenilla, Maria I; Ruiz, Cesar; Soto-Insuga, Víctor; Laguna, Laura Toledo Bravo de; Turón-Viñas, Eulàlia; Vázquez-López, M; Villar-Vera, Cristina

doi:10.1016/s1474-4422(19)30488-0

Cited by 237 publications

(392 citation statements)

References 29 publications

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“…Hemiparesis, visual field involvement, and signs of encephalopathy may be associated with large cerebral hemispheric lesions in 16–32% of AQP4-seropositive children ( 6 , 18 ). In a recently published pediatric cohort of 116 MOG-IgG seropositive patients, 4/6 children with an NMOSD phenotype had associated brain MRI findings, and 68 additional patients showed an encephalopathic presentation, including 64 with acute disseminated encephalomyelitis (ADEM) features and 22 satisfying criteria for autoimmune encephalitis without ADEM features ( 34 ).…”

Section: Review Of Current Knowledgementioning

confidence: 99%

Pediatric NMOSD: A Review and Position Statement on Approach to Work-Up and Diagnosis

Tenembaum

Yeh

2020

Front. Pediatr.

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Neuromyelitis Optica Spectrum Disorder (NMOSD) is an inflammatory demyelinating disease of the central nervous system (CNS) primarily affecting the optic nerves and spinal cord, but also involving other regions of the CNS including the area postrema, periaqueductal gray matter, and hypothalamus. Knowledge related to pediatric manifestations of NMOSD has grown in recent years, particularly in light of newer information regarding the importance of not only antibodies to aquaporin 4 (AQP4-IgG) but also myelin oligodendrocyte glycoprotein (MOG-IgG) in children manifesting clinically with this syndrome. In this review, we describe the current state of the knowledge related to clinical manifestations, diagnosis, and chronic therapies for children with NMOSD, with emphasis on literature that has been published in the last 5 years. Following the review, we propose recommendations for the assessment/follow up clinical care, and treatment of this population.

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Section: Review Of Current Knowledgementioning

confidence: 99%

Pediatric NMOSD: A Review and Position Statement on Approach to Work-Up and Diagnosis

Tenembaum

Yeh

2020

Front. Pediatr.

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“…25,26 Recently, Armangue et al showed that the spectrum of MOG-SD is wider than previously reported including children with encephalitis. 27 In their large cohort of children with ADS and encephalitis, they found that 22/296 children with definite or possible AE harbored MOG abs.…”

Section: Discussionmentioning

confidence: 99%

Clinical and imaging features of children with autoimmune encephalitis and MOG antibodies

Wegener-Panzer

Cleaveland

Wendel

et al. 2020

Neurol Neuroimmunol Neuroinflamm

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ObjectiveTo describe the presentations, radiologic features, and outcomes of children with autoimmune encephalitis associated with myelin oligodendrocyte glycoprotein antibodies (MOG abs).MethodsIdentification of children fulfilling the diagnostic criteria for possible autoimmune encephalitis (AE) and testing positive for serum MOG abs. Chart review and comprehensive analysis of serum MOG abs using live cell assays and rat brain immunohistochemistry.ResultsTen children (4 girls, 6 boys) with AE and serum MOG abs were identified. The median age at onset was 8.0 years (range: 4–16 years). Children presented with a combination of encephalopathy (10/10), headache (7/10), focal neurologic signs (7/10), or seizures (6/10). CSF pleocytosis was common (9/10, median 80 white cell count/μL, range: 21–256). Imaging showed cortical and deep gray matter involvement in all in addition to juxtacortical signal alterations in 6/10 children. No involvement of other white matter structures or contrast enhancement was noted. MOG abs were detected in all children (median titer 1:640; range: 1:320–1:10,540). Nine children had a favorable outcome at discharge (modified Rankin scale of < 2). Five of 10 children had up to 3 additional demyelinating relapses associated with persisting MOG abs. One child had NMDA receptor (NMDAR) abs at initial presentation. A second child had a third demyelinating episode with MOG abs with overlapping NMDAR encephalitis.DiscussionAE associated with serum MOG abs represents a distinct form of autoantibody-mediated encephalitis in children. We therefore recommend including MOG abs testing in the workup of children with suspected AE.

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“…Recurrent ADEM or ADEM associated with recurrent optic neuritis (52,53) are especially suggestive of MOGAD. Importantly, in children with clinical syndrome of encephalitis, MOGAD diagnosis is possible even when MRI findings are not compatible with ADEM-for example, exclusive cortical or symmetric thalamic/basal ganglia involvement, or even normal MRI (54).…”

Section: Acute Disseminated Encephalomyelitis (Adem) and Other Cerebrmentioning

confidence: 99%

The Expanding Clinical Spectrum of Myelin Oligodendrocyte Glycoprotein (MOG) Antibody Associated Disease in Children and Adults

Parrotta¹,

Kister

2020

Front. Neurol.

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Associations of paediatric demyelinating and encephalitic syndromes with myelin oligodendrocyte glycoprotein antibodies: a multicentre observational study

Cited by 237 publications

References 29 publications

Pediatric NMOSD: A Review and Position Statement on Approach to Work-Up and Diagnosis

Pediatric NMOSD: A Review and Position Statement on Approach to Work-Up and Diagnosis

Clinical and imaging features of children with autoimmune encephalitis and MOG antibodies

The Expanding Clinical Spectrum of Myelin Oligodendrocyte Glycoprotein (MOG) Antibody Associated Disease in Children and Adults

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