Abstract：Objective To study the clinical features and treatment outcome of children with myelin oligodendrocyte glycoprotein (MOG) antibody-associated disorders (MOGAD). Methods A retrospective analysis was performed for the clinical data of 28 children with MOGAD (with 38 demyelinating episodes). Results Among the disease spectrums of 28 children with MOGAD, optic neuritis was the most common (12 cases, 43%), followed by acute disseminated encephalomyelitis (9 cases, 32%). Among the 38 demyelinating episodes in the 28 children, there were 29 cases (76%) of lesions in the acute stage on head magnetic resonance imaging (MRI), and most of these lesions were extensive or isolated subcortical white matter lesions. A total of 24 cases of spinal MRI results in the acute stage were recorded, among which there were 11 cases (46%) of spinal lesions. MRI abnormalities of the optic nerve were found in 18 cases of optic neuritis in the acute stage. Of the 28 children, 20 (71%) had an increase in white blood cell count in cerebrospinal fluid, with lymphocytes as the most common type of cells, and 3 children had an increase in protein. The titer of serum MOG antibody was 1:10-1:320 in the 28 children. All 28 children were administered with glucocorticoids, along with immunoglobulin in 18 children. The symptoms of 26 children (93%) were alleviated during follow-up, and only 2 children had neurological sequela of the optic function. Conclusions The clinical manifestations are diverse in children with MOGAD. Immunotherapy is effective and most children have a good prognosis.
ZHENG Ping,ZHANG Jian-Zhao,SUN Jing et al. Clinical features of children with myelin oligodendrocyte glycoprotein antibodyassociated disorders[J]. CJCP, 2020, 22(4): 368-373.
Reindl M, Waters P. Myelin oligodendrocyte glycoprotein antibodies in neurological disease[J]. Nat Rev Neurol, 2019, 15(2):89-102.
[2]
Dubey D, Pittock SJ, Krecke KN, et al. Clinical, radiologic, and prognostic features of myelitis associated with myelin oligodendrocyte glycoprotein autoantibody[J]. JAMA Neurol, 2019, 76(3):301-309.
[3]
Jurynczyk M, Geraldes R, Probert F, et al. Distinct brain imaging characteristics of autoantibody-mediated CNS conditions and multiple sclerosis[J]. Brain, 2017, 140(3):617-627.
[4]
Jarius S, Paul F, Aktas O, et al. MOG encephalomyelitis:international recommendations on diagnosis and antibody testing[J]. J Neuroinflammation, 2018, 15(1):134.
[5]
Ramanathan S, Dale RC, Brilot F. Anti-MOG antibody:the history, clinical phenotype, and pathogenicity of a serum biomarker for demyelination[J]. Autoimmun Rev, 2016, 15(4):307-324.
[6]
Nagabushana D, Shah R, Pendharkar H, et al. MOG antibody seropositive aseptic meningitis:a new clinical phenotype[J]. J Neuroimmunol, 2019, 333:476960.
[7]
Jurynczyk M, Messina S, Woodhall MR, et al. Clinical presentation and prognosis in MOG-antibody disease:a UK study[J]. Brain, 2017, 140(12):3128-3138.
[8]
Cobo-Calvo A, Vukusic S, Marignier R. Clinical spectrum of central nervous system myelin oligodendrocyte glycoprotein autoimmunity in adults[J]. Curr Opin Neurol, 2019, 32(3):459-466.
[9]
Hennes EM, Baumann M, Lechner C, et al. MOG spectrum disorders and role of MOG-antibodies in clinical practice[J]. Neuropediatrics, 2018, 49(1):3-11.
[10]
Ogawa R, Nakashima I, Takahashi T, et al. MOG antibodypositive, benign, unilateral, cerebral cortical encephalitis with epilepsy[J]. Neurol Neuroimmunol Neuroinflamm, 2017, 4(2):e322.
[11]
Ikeda T, Yamada K, Ogawa R, et al. The pathological features of MOG antibody-positive cerebral cortical encephalitis as a new spectrum associated with MOG antibodies:a case report[J]. J Neurol Sci, 2018, 392:113-115.
Hennes EM, Baumann M, Schanda K, et al. Prognostic relevance of MOG antibodies in children with an acquired demyelinating syndrome[J]. Neurology, 2017, 89(9):900-908.
[14]
Kim SM, Woodhall MR, Kim JS, et al. Antibodies to MOG in adults with inflammatory demyelinating disease of the CNS[J]. Neurol Neuroimmunol Neuroinflamm, 2015, 2(6):e163.
[15]
Cobo-Calvo A, Ruiz A, Maillart E, et al. Clinical spectrum and prognostic value of CNS MOG autoimmunity in adults:the MOGADOR study[J]. Neurology, 2018, 90(21):e1858-e1869.
[16]
Hyun JW, Woodhall MR, Kim SH, et al. Longitudinal analysis of myelin oligodendrocyte glycoprotein antibodies in CNS inflammatory diseases[J]. J Neurol Neurosurg Psychiatry, 2017, 88(10):811-817.
[17]
López-Chiriboga AS, Majed M, Fryer J, et al. Association of MOG-IgG serostatus with relapse after acute disseminated encephalomyelitis and proposed diagnostic criteria for MOGIgG-associated disorders[J]. JAMA Neurol, 2018, 75(11):1355-1363.
[18]
Spadaro M, Gerdes LA, Mayer MC, et al. Histopathology and clinical course of MOG-antibody-associated encephalomyelitis[J]. Ann Clin Transl Neurol, 2015, 2(3):295-301.
[19]
Zhou Y, Jia X, Yang H, et al. Myelin oligodendrocyte glycoprotein antibody-associated demyelination:comparison between onset phenotypes[J]. Eur J Neurol, 2019, 26(1):175-183.
[20]
Jarius S, Ruprecht K, Kleiter I, et al. 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]. J Neuroinflammation, 2016, 13(1):280.
[21]
Polat İ, Yiş U, Karaoğlu P, et al. Myelin oligodendrocyte glycoprotein antibody persistency in a steroid-dependent ADEM case[J]. Pediatrics, 2016, 137(5). pii:e20151958.
[22]
Hacohen Y, Wong YY, Lechner C, et al. Disease course and treatment responses in children with relapsing myelin oligodendrocyte glycoprotein antibody-associated disease[J]. JAMA Neurol, 2018, 75(4):478-487.
