Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica

Rostásy, Kevin; Mader, Simone; Hennes, Eva-Maria; Schanda, Kathrin; Gredler, Viktoria; Guenther, Armin; Blaschek, Astrid; Korenke, Christoph; Pritsch, Martin; Pohl, Daniela; Maier, Oskar; Kuchukhidze, Giorgi; Brunner‐Krainz, Michaela; Berger, Thomas; Reindl, Markus

doi:10.1177/1352458512470310

Cited by 152 publications

(148 citation statements)

References 24 publications

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“…Anti-MOG antibodies occur more frequently in children than in adults and have been described (using different assays which, as noted above, may affect findings) in pediatric patients with a variety of autoimmune demyelinating disease phenotypes, including those of ADEM, MS, 32,[35][36][37] relapsing ADEM, relapsing ON, 38,39 relapsing ADEM-ON, 40 and relapsing ON-longitudinally extensive TM (NMO-like). 36,37 The normal function of MOG, which resides at the cell surface of oligodendrocytes, is not well understood.…”

Section: Cns-directed Antibodies In Pediatricmentioning

confidence: 99%

Immunopathophysiology of pediatric CNS inflammatory demyelinating diseases

et al. 2016

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Elucidating pathophysiologic mechanisms underlying the spectrum of pediatric-onset CNS demyelinating diseases, particularly those that may distinguish multiple sclerosis (MS) from other entities, promises to both improve diagnostics and guide more-informed therapeutic decisions. Observations that pediatric-and adult-onset MS share the same genetic and environmental risk factors support the view that these conditions represent essentially the same illness manifesting at different ages. Nonetheless, special consideration must be given when CNS inflammation manifests in early life, at a time when multiple organs (including immune and nervous systems) are actively maturing. CSF analysis in pediatric-onset MS points to chronic CNS inflammation, supported by observations from limited pathologic material available for study. Emerging results implicate abnormalities in both effector and regulatory T cell subsets, and potentially immune senescence, in children with MS. Although CNS-directed antibodies (including antibodies recognizing myelin antigens; Kir4.1) can be documented in pediatric-onset MS, their pathophysiologic significance (as in adults) remains unclear. This is in contrast to the presence of serum and/or CSF antibodies recognizing aquaporin-4, which, when measured using validated cell-based assays, supports the diagnosis of a neuromyelitis optica spectrum disorder, distinct from MS. Presence of anti-myelin oligodendrocyte glycoprotein antibodies documented with similar cell-based assays may also be associated with pathophysiologically distinct disease phenotypes in children. The substantial impact of pediatriconset MS on normal brain development and function underscores the importance of elucidating both the immunobiology and neurobiology of disease. Ongoing efforts are aimed at developing and validating biological measures that define pathophysiologically distinct monophasic and chronic forms of pediatric CNS demyelination. Neurology ® 2016;87 (Suppl 2):S12-S19 GLOSSARY ADEM 5 acute disseminated encephalomyelitis; AQP4 5 aquaporin-4; BBB 5 blood-brain barrier; CBA 5 cell-based assay; EDSS 5 Expanded Disability Status Scale; MBP 5 myelin basic protein; MOG 5 myelin oligodendrocyte glycoprotein; MS 5 multiple sclerosis; NMO 5 neuromyelitis optica; NMOSD 5 NMO spectrum disorder; OCB 5 oligoclonal band; ON 5 optic neuritis; OPC 5 oligodendrocyte precursor cell; TM 5 transverse myelitis.There is presently limited insight into the pathophysiologic mechanisms underlying childhoodonset inflammatory demyelinating diseases. Because these disorders occur in the context of the developing immune and nervous systems, understanding the implications to both disease mechanisms and efficacy and safety profiles of potential therapeutics will help guide optimal management approaches for these children and adolescents. Elucidating the biology of pediatric-onset multiple sclerosis (MS) may provide key insights into earliest targets and processes involved in disease pathogenesis as well as into the broader spectrum of CNS...

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Section: Cns-directed Antibodies In Pediatricmentioning

confidence: 99%

Immunopathophysiology of pediatric CNS inflammatory demyelinating diseases

et al. 2016

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“…[14][15][16][17][18] In contrast, the clinical relevance of MOG antibodies in adults is unclear, as only a minority of patients with MS and NMO/NMOSD are seropositive. 16,[18][19][20][21][22][23][24] Herein, we provide evidence that MOG antibodies are a clinical biomarker of bilateral and/ or recurrent optic neuritis (BON) in adults and describe the characteristic clinical course, response to therapy, and visual outcomes of this condition.…”

mentioning

confidence: 99%

Antibodies to myelin oligodendrocyte glycoprotein in bilateral and recurrent optic neuritis

Ramanathan

Reddel

Henderson

et al. 2014

Journal of Neuroimmunology

110

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“…6 It was shown that some children with ADEM had high titers of MOG antibodies (≥1:160) that rapidly decreased to the undetectable levels after the first attack. 7,8 Strikingly, persistence with fluctuations of anti-MOG IgG was noted in children with MS. 7 MOG antibodies rapidly declined in patients who completely recovered from ADEM. Decreasing MOG antibody titers show favorable clinical outcome in patients with ADEM.…”

Section: Figurementioning

confidence: 98%

“…Rostasy et al 8 showed that the presence of high-titer anti-MOG IgG in a subgroup of patients with recurrent ON as compared with children with monophasic ON. In their study, the patients with recurrent attacks needed to be treated with immunomodulators, including AZA, rituximab, and IVIG.…”

Section: Figurementioning

confidence: 99%

“…It was mentioned that the children with recurrent ON episodes have persistent high-titer MOG antibodies and the children with anti-MOG IgG-positive NMO had more benign disease course with response to milder immunomodulation theraphies. 8 Sato et al 10 reported that NMO spectrum disorders with positive MOG and negative AQP4 antibodies more often have single attack and better functional recovery. According to recent reports, it cannot be said with certainly that antibody positivity predicts the subtype of demyelinating disease.…”

Section: Figurementioning

confidence: 99%

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Myelin Oligodendrocyte Glycoprotein Antibody Persistency in a Steroid-Dependent ADEM Case

Polat¹,

Yış²,

Karaoğlu³

et al. 2016

Pediatrics

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Myelin oligodendrocyte glycoprotein (MOG) is a candidate target antigen in demyelinating central nervous system diseases, including acute disseminated encephalomyelitis (ADEM), neuromyelitis optica, and multiple sclerosis. It may give prognostic information regarding monophasic or recurrent course of the disease. MOG antibodies have been shown to be positive in high titers during the first episode of ADEM with rapidly decreasing to undetectable limits after recovery. However, persistent MOG antibodies are considered as a predicting factor for multiple sclerosis, optic neuritis relapses, and incomplete recovery of ADEM. Here we report a unique case with persistent MOG antibodies presented with multiphasic ADEM-like attacks. A 6-year-old girl was consulted with encephalopathy, gait disturbance, and oculomotor nerve palsy. Periventricular white matter lesions were seen on cranial magnetic resonance imaging studies. ADEM was diagnosed and treated with steroid. During follow-up, she experienced repeated episodes after steroid therapy termination. We were able to search MOG antibody at the ninth attack. The positivity of this antibody remained. It was thought to be associated with steroid-dependent course, and azathioprine and intravenous human immunoglobulin treatment were added. Patients with persistent MOG antibodies may benefit from addition of immunosuppressant agents, which may decrease the number of attacks.

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Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica

Abstract: Pediatric patients presenting with clinical symptoms and MRI findings highly suggestive of NMO but with high and persisting MOG-IgG antibody titers are most likely to represent a distinct subgroup of acute demyelinating diseases with important clinical and therapeutic implications.

Cited by 152 publications

References 24 publications

Immunopathophysiology of pediatric CNS inflammatory demyelinating diseases

Immunopathophysiology of pediatric CNS inflammatory demyelinating diseases

Antibodies to myelin oligodendrocyte glycoprotein in bilateral and recurrent optic neuritis

Myelin Oligodendrocyte Glycoprotein Antibody Persistency in a Steroid-Dependent ADEM Case

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