Refractory MOG-Associated Demyelinating Disease in a Pediatric Patient

Kroenke, Eve; Ankar, Alex; Shukla, Nikita

doi:10.1177/2329048x221079093

Cited by 2 publications

(3 citation statements)

References 11 publications

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“…Some retrospective studies suggest that children with relapsing MOGAD refractory to rituximab and IVIG may respond to tocilizumab. [ 78 87 ]…”

Section: Onoclonal a Ntibody T ...mentioning

confidence: 99%

Monoclonal antibody therapies for aquaporin-4-immunoglobulin G-positive neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease

Tisavipat,

Juan,

Chen

2023

Saudi Journal of Ophthalmology

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Monoclonal antibody therapies mark the new era of targeted treatment for relapse prevention in aquaporin-4 (AQP4)-immunoglobulin G (IgG)-positive neuromyelitis optica spectrum disorder (AQP4-IgG+NMOSD). For over a decade, rituximab, an anti-CD20 B-cell-depleting agent, had been the most effectiveness treatment for AQP4-IgG+NMOSD. Tocilizumab, an anti-interleukin-6 receptor, was also observed to be effective. In 2019, several randomized, placebo-controlled trials were completed that demonstrated the remarkable efficacy of eculizumab (anti-C5 complement inhibitor), inebilizumab (anti-CD19 B-cell-depleting agent), and satralizumab (anti-interleukin-6 receptor), leading to the Food and Drug Administration (FDA) approval of specific treatments for AQP4-IgG+NMOSD for the first time. Most recently, ravulizumab (anti-C5 complement inhibitor) was also shown to be highly efficacious in an open-label, external-controlled trial. Although only some patients with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) warrant immunotherapy, there is currently no FDA-approved treatment for relapse prevention in MOGAD. Observational studies showed that tocilizumab was associated with a decrease in relapses, whereas rituximab seemed to have less robust effectiveness in MOGAD compared to AQP4-IgG+NMOSD. Herein, we review the evidence on the efficacy and safety of each monoclonal antibody therapy used in AQP4-IgG+NMOSD and MOGAD, including special considerations in children and women of childbearing potential.

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“…Some retrospective studies suggest that children with relapsing MOGAD refractory to rituximab and IVIG may respond to tocilizumab. [ 78 87 ]…”

Section: Onoclonal a Ntibody T ...mentioning

confidence: 99%

Monoclonal antibody therapies for aquaporin-4-immunoglobulin G-positive neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease

Tisavipat,

Juan,

Chen

2023

Saudi Journal of Ophthalmology

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“…The most common MOGA-associated encephalitis, MOGA-associated optic neuritis, [1] MOGA-associated encephalomyelitis, and others. [2,3] MOG is a myelin glycoprotein expressed on the outermost of the myelin sheath and oligodendrocyte membranes in the CNS, despite the reality that MOG is an extremely minor component of the myelin sheath, and it is been recognized as a putative candidate autoantigen and autoantibody target in demyelination. [4] MO and NMK contributed equally to this work.…”

Section: Introductionmentioning

confidence: 99%

“…The most common MOGA-associated encephalitis, MOGA-associated optic neuritis, [1] MOGA-associated encephalomyelitis, and others. [2,3]…”

Section: Introductionmentioning

confidence: 99%

Pediatric myelin oligodendrocyte glycoprotein antibody associated disease—Asymmetric papilledema and elevated ICP are two of the chameleons: A case report

et al. 2023

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Background: Myelin oligodendrocyte glycoprotein antibody (MOGA) associated diseases are inflammatory immune-mediated demyelinating disorders with relapse potential involving the central nervous system. Multiple unusual clinical manifestations of those disorders were reported, making treatment decisions difficult.Case presentation: A healthy 12-year-old obese boy presented with headache and bilateral asymmetric papilledema. The patient had a negative medical history. His neurological and general examinations were unremarkable, his initial magnetic resonance imaging showed elevated intracranial pressure (ICP) only. A lumbar puncture revealed increased opening pressure and pleocytosis. The MOGA titer was 1:320. He needed acetazolamide and steroid therapy. After 2 months of medication, weight loss, exercise, the patient symptoms significantly improved, papilledema resolved, and visual function improved.Conclusion: MOGA-associated disorders have a variety of clinical features, so a high index of suspicion is required for their diagnosis. Papilledema and an elevated ICP are 2 of the chameleons of MOGA-associated disorders. MOGA test may be useful in patients with elevated ICP and inflammatory cerebrospinal fluid profiles. An investigation of the possible association between those disorders and high ICP is warranted.Abbreviations: ADEM = acute disseminated encephalomyelitis, CNS = central nervous system, CSF = cerebrospinal fluid, ICP = intracranial pressure, IIH = idiopathic intracranial hypertension, MOG = myelin oligodendrocyte glycoprotein, MOGA = myelin oligodendrocyte glycoprotein antibody, MRI = magnetic resonance imaging.

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Refractory MOG-Associated Demyelinating Disease in a Pediatric Patient

Cited by 2 publications

References 11 publications

Monoclonal antibody therapies for aquaporin-4-immunoglobulin G-positive neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease

Monoclonal antibody therapies for aquaporin-4-immunoglobulin G-positive neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease

Pediatric myelin oligodendrocyte glycoprotein antibody associated disease—Asymmetric papilledema and elevated ICP are two of the chameleons: A case report

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