- •Anti-NMDAR encephalitis and MOG ab related disease can occur independently or simultaneously.
- •Children with typical anti-NMDAR encephalitis can combine with positive MOG ab only.
- •More clinical overlaps exist between NMDAR ab(+)/MOG ab(+) group and NMDAR ab(+)/MOG ab(-) group.
- •More MRI overlaps exist between NMDAR ab(+)/MOG ab(+) group and MOG ab(+)/NMDAR ab(-) group.
- •MOG ab is associated with relapse, and NMDAR ab is associated with neurological sequelae.
To analyze the clinical features in children with anti-NMDAR encephalitis combined with myelin oligodendrocyte glycoprotein antibody (MOG ab).
Clinical data of 7 children with anti-NMDAR encephalitis combined with MOG ab(+) were collected in Guangzhou Women and Children's Medical Center from January, 2016 to June, 2019. Children with NMDAR ab(+)/MOG ab(-) and MOG ab(+)/NMDAR ab(-) were randomly selected as controls.
Onset age was 6.0 (IQR 5.0–7.0) years old, male to female was 2:5. Prominent symptoms include abnormal mental behavior (7/7), sleep disorder (6/7), speech disorder (6/7), involuntary movement (4/7) and paralysis (4/7). There were significant differences between NMDAR ab(+)/MOG ab(+) group versus MOG ab(+)/NMDAR ab(-) and NMDAR ab(+)/MOG ab(-) group versus MOG ab(+)/NMDAR ab(-) group (P< 0.0167, Fisher exact tests) in abnormal mental behavior, sleep disorder, speech disorder and involuntary movement. 1 case developed anti-NMDAR encephalitis 1 year after recovery from MOG ab related acute disseminated encephalomyelitis (ADEM). 4 cases developed anti-NMDAR encephalitis and MOG ab related ADEM simultaneously, with 2 cases relapsed. 2 cases were anti-NMDAR encephalitis with only MOG ab positive. In terms of MRI, there were differences in subcortical white matter, basal ganglia and brainstem (P < 0.0167, Fisher exact tests) between NMDAR ab(+)/MOG ab(+) group versus NMDAR ab(+)/MOG ab(-) (P < 0.0001) and NMDAR ab(+)/MOG ab(-) group versus MOG ab(+)/NMDAR ab(-) group(P<0.0001). There were significant differences in MOG antibody titer (Z = −=2.03, P = 0.042) and duration (Z = −1.97, P = 0.049) between relapsed and non-relapsed patients. 3 cases had neurological sequelae. The differences of NMDAR antibody titer (Z = −2.22, P = 0.026) and duration (Z = −2.18, P = 0.029) were significant between patients with and without neurological sequelae.
NMDAR and MOG antibodies can coexist in children with autoimmune encephalitis. Double antibody positive subjects had more overlaps in clinical manifestations with NMDAR encephalitis, and more overlaps in MRI changes with MOG ab related disease. Higher persistent MOG antibody titer may indicate recurrence, while higher persistent NMDAR antibodies titer may cause neurological sequelae.
Abbreviations:Ab (antibody), ADEM (acute disseminated encephalomyelitis), AQP4 (aquaporin-4), CNS (central nervous system), CSF (cerebrospinal fluid), EEG (electroencephalogram), IVIG (intravenous immunoglobulin), IVMP (intravenous methylprednisolone), MOG (myelin oligodendrocyte glycoprotein), MRI (magnetic resonance imaging), mRS (modified Rankin score), MDEM (multiphasic disseminated encephalomyelitis), NMDAR (N-methyl-d-aspartate receptor), NMOSD (optic myelomyelitis spectrum disorder), Pro (protein), WBC (white blood cell)
To read this article in full you will need to make a payment
Purchase one-time access:Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
One-time access price info
- For academic or personal research use, select 'Academic and Personal'
- For corporate R&D use, select 'Corporate R&D Professionals'
Subscribe:Subscribe to Multiple Sclerosis and Related Disorders
Already a print subscriber? Claim online access
Already an online subscriber? Sign in
Register: Create an account
Institutional Access: Sign in to ScienceDirect
- Different features between pediatric-onset and adult-onset patients who are seropositive for MOG-IgG: a multicenter study in South China.J. Neuroimmunol. 2018; 321: 83-91https://doi.org/10.1016/j.jneuroim.2018.05.014
- Anti-NMDA-receptor encephalitis: case series and analysis of the effects of antibodies.Lancet Neurol. 2008; 7: 1091-1098https://doi.org/10.1016/S1474-4422(08)70224-2
- Paraneoplastic anti-N-methyl-d-aspartate receptor encephalitis associated with ovarian teratoma.Ann. Neuro.l. 2007; 61: 25-36https://doi.org/10.1002/ana.21050
- Comparison of myelin oligodendrocyte glycoprotein (MOG)-antibody disease and AQP4-IgG-positive neuromyelitis optica spectrum disorder (NMOSD) when they co-exist with anti-NMDA (N-methyl-d-aspartate) receptor encephalitis.Mult. Scler. Relat. Disord. 2018; 20: 144-152https://doi.org/10.1016/j.msard.2018.01.007
- A clinical approach to diagnosis of autoimmune encephalitis.Lancet Neurol. 2016; 15: 391-404https://doi.org/10.1016/S1474-4422(15)00401-9
- Antibody titres at diagnosis and during follow-up of anti-NMDA receptor encephalitis: a retrospective study.Lancet Neurol. 2014; 13: 167-177https://doi.org/10.1016/S1474-4422(13)70282-5
- MOG encephalomyelitis: international recommendations on diagnosis and antibody testing.J. Neuroinflamm. 2018; 15: 134https://doi.org/10.1186/s12974-018-1144-2
- MOG-IgG in nmo and related disorders: a multicenter study of 50 patients. Part 2: epidemiology, clinical presentation, radiological and laboratory features, treatment responses, and long-term outcome.J. Neuroinflamm. 2016; 13: 280https://doi.org/10.1186/s12974-016-0718-0
- International Pediatric Multiple Sclerosis Study Group criteria for pediatric multiple sclerosis and immune-mediated central nervous system demyelinating disorders: revisions to the 2007 definitions.Mult. Scler. 2013; 19: 1261-1267https://doi.org/10.1177/1352458513484547
- Acute psychosis in a pregnant patient with Graves' hyperthyroidism and anti-NMDA receptor encephalitis.BMJ Case Rep. 2015; (2015)https://doi.org/10.1136/bcr-2014-208052
- A case of anti-myelin oligodendrocyte glycoprotein (MOG) and anti-N-methyl-d-aspartate (NMDA) receptor antibody-positive encephalitis with optic neuritis.Rinsho Shinkeigaku. 2018; 58: 636-641https://doi.org/10.5692/clinicalneurol.cn-001194
- Myelin oligodendrocyte glycoprotein: deciphering a target in inflammatory demyelinating diseases.Front. Immunol. 2017; 8: 529https://doi.org/10.3389/fimmu.2017.00529
- Clinical manifestations, treatment outcomes, and prognostic factors of pediatric anti-NMDAR encephalitis in tertiary care hospitals: a multicenter retrospective/prospective cohort study.Brain Dev. 2019; 41: 436-442https://doi.org/10.1016/j.braindev.2018.12.009
- Anti-NMDAR encephalitis followed by seropositive neuromyelitis optica spectrum disorder: a case report and literature review.Clin. Neurol. Neurosurg. 2017; 155: 75-82https://doi.org/10.1016/j.clineuro.2017.02.016
- Co-occurrence of anti-N-methyl-d-aspartate receptor encephalitis and anti-myelin oligodendrocyte glycoprotein inflammatory demyelinating diseases: a clinical phenomenon to be taken seriously.Front. Neurol. 2019; 10: 1271https://doi.org/10.3389/fneur.2019.01271
- A case of recurrent MOG antibody positive bilateral optic neuritis and anti-NMDAR encephalitis: different biological evolution of the two associated antibodies.J. Neuroimmunol. 2019; 328: 86-88https://doi.org/10.1016/j.jneuroim.2018.12.003
- A rare concurrence: antibodies against myelin oligodendrocyte glycoprotein and N-methyl-d-aspartate receptor in a child.Mult. Scler. Relat. Disord. 2019; 28: 101-103https://doi.org/10.1016/j.msard.2018.12.017
- N-methyl-d-aspartate receptor antibody encephalitis: a concise review of the disorder, diagnosis, and management.ACS Chem. Neurosci. 2019; 10: 132-142https://doi.org/10.1021/acschemneuro.8b00304
- Overlapping demyelinating syndromes and anti–N-methyl-d-aspartate receptor encephalitis.Ann. Neurol. 2014; 75: 411-428https://doi.org/10.1002/ana.24117
- Encephalitis is an important clinical component of myelin oligodendrocyte glycoprotein antibody associated demyelination: a single-center cohort study in Shanghai, China.Eur. J. Neurol. 2019; 26: 168-174https://doi.org/10.1111/ene.13790
- Neurological update: MOG antibody disease.J. Neurol. 2019; 266: 1280-1286https://doi.org/10.1007/s00415-018-9122-2
- Cerebral cortical encephalitis followed by recurrent CNS demyelination in a patient with concomitant anti-MOG and anti-NMDA receptor antibodies.Mult. Scler. Relat. Disord. 2017; 18: 90-92https://doi.org/10.1016/j.msard.2017.09.023
Published online: February 22, 2020
Accepted: February 21, 2020
Received in revised form: February 7, 2020
Received: December 8, 2019
© 2020 Elsevier B.V. All rights reserved.