Highlights
- •Through analysis of thirty pediatric patients diagnosed with MOGAD and treated with MMF for >1 year, our study further identified the efficacy and safety of MMF in treating pediatric MOGAD.
- •We explored risk factors which could predict recurrence during MMF treatment of MOGAD in our study, but no correlation between clinical features and relapse was found.
- •We reported some cases with escalation or de-escalation therapy in MOGAD cohort. Due to the small number of cases, no consistent conclusions can be drawn. Further studies are needed in this respect.
Abstract
Objective
To evaluate the efficacy of mycophenolate mofetil (MMF) in the treatment of childhood
MOG-IgG-associated disorder (MOGAD).
Methods
Thirty patients diagnosed with relapsing MOGAD and treated with MMF for >1 year from
a childhood MOGAD ambispective cohort were included in the study. The clinical characteristics,
therapeutic regimen, side effects, annualized relapse rate (ARR), and Expanded Disability
Status Scale (EDSS) scores of these patients were evaluated.
Results
The median age of disease onset was 7.05 (2.50–12.75) years. The male to female ratio
was 1:1.31. All patients used MMF as first-line maintenance treatment. The median
time to add MMF from disease onset was 1.08 (0.25−5.00) year. The median number of
attacks before MMF initiation was 2 (2 − 8). The median duration of MMF therapy was
2.13 (1.00−3.58) years. Twenty (66.67%) patients did not experience further attacks
during MMF therapy. The Kaplan–Meier curves showed a 3-year relapse-free rate of 59.8%
(95% CI, 36.62−76.88%). ARR decreased during MMF therapy (0 (0 − 1.72) vs. 1.25 (0.60−4.00);
P < 0.05). EDSS stabilized during MMF therapy (1.0 (0 − 2.0) vs. 0 (0 − 2.0); P = 0.206). None of the patients stopped the use of MMF due to intolerable side effects.
Onset age, sex, phenotype of the first attack, ARR before MMF, MOG-IgG titers, and
combined long-term prednisone (prednisone <10 mg daily for patients >40 kg or <5 mg
daily for patients ≤40 kg longer than 6 months) did not predict recurrence during
MMF therapy in univariate analysis.
Conclusions
MMF was effective and safe for treating childhood MOGAD. No clinical feature that
could predict efficacy of MMF was found in pediatric patients with MOGAD.
Keywords
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 accessOne-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 DisordersAlready a print subscriber? Claim online access
Already an online subscriber? Sign in
Register: Create an account
Institutional Access: Sign in to ScienceDirect
References
- E.U. paediatric MOG consortium consensus: part 5 - Treatment of paediatric myelin oligodendrocyte glycoprotein antibody-associated disorders.Eur. J. Paediatr. Neurol. 2020; 29: 41-53
- Steroid-sparing maintenance immunotherapy for MOG-IgG associated disorder.Neurology. 2020; 95: e111-e120
- Risk factors for academic difficulties in children with myelin oligodendrocyte glycoprotein antibody-associated acute demyelinating syndromes.Dev. Med. Child. Neurol. 2020; 62: 1075-1081
- Comparison of the response to rituximab between myelin oligodendrocyte glycoprotein and aquaporin-4 antibody diseases.Ann. Neurol. 2020; 87: 256-266
- Interleukin-6 inhibition with tocilizumab for relapsing MOG-IgG associated disorder (MOGAD): a case-series and review.Mult. Scler. Relat. Disord. 2021; 48102696
- Disease course and treatment responses in children with relapsing myelin oligodendrocyte glycoprotein antibody-associated disease.JAMA Neurol. 2018; 75: 478-487
- Prognostic relevance of MOG antibodies in children with an acquired demyelinating syndrome.Neurology. 2017; 89: 900-908
- Neuromyelitis optica.Nat. Rev. Dis. Primer. 2020; 6: 85
- Clinical presentation and prognosis in MOG-antibody disease: a UK study.Brain. 2017; 140: 3128-3138
- Interleukin-6 receptor blockade in treatment-refractory MOG-IgG-associated disease and neuromyelitis optica spectrum disorders.Neurol. Neuroimmunol. Neuroinflamm. 2021; 9: e1100
- MOG-IgG1 and co-existence of neuronal autoantibodies.Mult. Scler. 2021; 27: 1175-1186
- Long-term efficacy of mycophenolate mofetil in myelin oligodendrocyte glycoprotein antibody-associated disorders: a prospective study.Neurol. Neuroimmunol. Neuroinflamm. 2020; 7: e705
- Safety and efficacy of rituximab for relapse prevention in myelin oligodendrocyte glycoprotein immunoglobulin G (MOG-IgG)-associated disorders (MOGAD): a systematic review and meta-analysis.J. Neuroimmunol. 2022; 364577812
- MOG antibody-positive, benign, unilateral, cerebral cortical encephalitis with epilepsy.Neurol. Neuroimmunol. Neuroinflamm. 2017; 4: e322
- Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination.J. Neurol. Neurosurg. Psychiatry. 2018; 89: 127-137
- Myelin oligodendrocyte glycoprotein antibody-associated disease: an immunopathological study.Brain. 2020; 143: 1431-1446
- Differential efficacy of mycophenolate mofetil in adults with relapsing myelin oligodendrocyte glycoprotein antibody-associated disorders.Mult. Scler. Relat. Disord. 2021; 53103035
- Pediatric anti-N-methyl-d-aspartate receptor encephalitis with MOG-Ab co-existence: relapse propensity and treatability.Mult. Scler. Relat. Disord. 2022; 58103447
- Treatment of MOG-IgG-associated disorder with rituximab: an international study of 121 patients.Mult. Scler. Relat. Disord. 2020; 44102251
- Noncommunicable chronic disease prevention should start from childhood.Pediatr. Investig. 2021; 5: 3-5
- Follow-up study on Chinese children with relapsing MOG-IgG-associated central nervous system demyelination.Mult. Scler. Relat. Disord. 2019; 28: 4-10
- Clinical features of anti-myelin oligodendrocyte glycoprotein antibody-associated diseases in children with cortical encephalitis.Zhonghua Yi Xue Za Zhi. 2020; 100: 1952-1955
Article info
Publication history
Published online: August 20, 2022
Accepted:
August 16,
2022
Received in revised form:
July 14,
2022
Received:
March 24,
2022
Identification
Copyright
© 2022 Elsevier B.V. All rights reserved.
