Retrospective analysis of children with myelin oligodendrocyte glycoprotein antibody-related disorders

Konuşkan, Bahadır; Yıldırım, Miraç; Göçmen, Rahşan; Okur, Tuncay Derya; Polat, İpek; Kılıç, Hüseyin; Saltık, Sema; Öztürk, Zeynep; Gücüyener, Kıvılcım; Altunbaşak, Şakìr; Çelik, Tamer; Köse, Gülşen; Yılmaz, Arzu; Kömür, Mustafa; Kayılıoğlu, Hülya; Anlar, Banu

doi:10.1016/j.msard.2018.07.022

Cited by 35 publications

(30 citation statements)

References 28 publications

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“…28 Clinically, patients with MOG antibody seropositive ADEM have a similarly heterogenous presentation with regard to neurologic disease, although rates of ON are reported to be higher both concurrently and as a relapsing phenotype. [27][28][29] Although seizures have been reported in patients with ADEM, it appears that MOG antibody-positive patients with ADEM may have increased rates of both seizure at presentation and long-term risk of post-ADEM epilepsy which is another distinguishing characteristic when compared with other MOG antibody spectrum disorders. 30 Diagnostically, children with MOG antibody-associated ADEM are reported to have an increased rate of large, bilateral, and widespread lesions including the brain and spinal cord.…”

Section: Mog Antibody and Ademmentioning

confidence: 99%

“…31 Interestingly, following initiation of immunotherapy lesions are more likely to resolve (or significantly decrease in size) on follow-up scans when compared with non-MOG antibodyassociated ADEM and other MOG antibody spectrum disorders. [26][27][28][29]31,32 The presence of MOG antibodies in ADEM poses a longterm monitoring challenge in patients, as monophasic and multiphasic forms have been reported. Monophasic illness accounts for the majority of ADEM with MOG antibodypositive cases and has been associated with male gender, younger age at diagnosis (<10 years), and pathology not involving the optic nerves.…”

Section: Mog Antibody and Ademmentioning

confidence: 99%

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Diagnostic Considerations in Acute Disseminated Encephalomyelitis and the Interface with MOG Antibody

Santoro

Chitnis

2019

Neuropediatrics

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Acute disseminated encephalomyelitis (ADEM) is a common yet clinically heterogenous syndrome characterized by encephalopathy, focal neurologic findings, and abnormal neuroimaging. Differentiating ADEM from other demyelinating disorders of childhood can be difficult and appropriate interpretation of the historical, clinical, and neurodiagnostic components of a patient's presentation is critical. Myelin oligodendrocyte glycoprotein (MOG) antibody-associated diseases are a recently recognized set of disorders, which include ADEM presentations, among other phenotypes. This review article discusses the clinical diagnosis, differential diagnosis, interpretation of data, and treatment/prognosis of this unique syndrome with distinctive review of the spectrum of MOG antibodies.

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Section: Mog Antibody and Ademmentioning

confidence: 99%

Section: Mog Antibody and Ademmentioning

confidence: 99%

Diagnostic Considerations in Acute Disseminated Encephalomyelitis and the Interface with MOG Antibody

Santoro

Chitnis

2019

Neuropediatrics

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“…21 Four retrospective studies have described treatment response to therapies used in children positive for anti-MOG antibodies. Three of these studies identified patients purely on MOG-antibody status and relapses [22][23][24][25] and one using a clinically-defined population. In the three studies focusing purely on anti-MOG antibody-positive patients, reduction in the relapse rate occurred using most therapies, including monthly IVIG, rituximab, mycophenolate mofetil, azathioprine, and steroids.…”

Section: Discussionmentioning

confidence: 99%

Rituximab in children with myelin oligodendrocyte glycoprotein antibody and relapsing neuroinflammatory disease

Albassam

Longoni

Yea

et al. 2019

Develop Med Child Neuro

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ABBREVIATIONS IVIGIntravenous immunoglobulin MOG Myelin oligodendrocyte glycoproteinThe aim of this study was to evaluate tolerability of and response to rituximab in children with myelin oligodendrocyte glycoprotein (MOG) antibody-positive relapsing neuroinflammatory disease. This was an observational study of prospectively collected data on 12 consecutive children (eight females, four males; median age at onset 10y 6mo [interquartile range {IQR} 7y 2mo-12y 5mo], median follow-up 2y 1mo [IQR 1y 7mo-2y 6mo]) with central nervous system inflammation and persistent serum MOG immunoglobulin G positivity more than 12 weeks after clinical presentation. Patients received a standardized rituximab treatment protocol. MOG antibody testing was performed following standardized cell-based methods. Median clinical follow-up after rituximab induction was 2 years (IQR 1y 7mo-2y 10mo). The relapse rate in the first 12 months posttreatment was 0 (IQR 0-0). After rituximab, two patients relapsed during B-cell suppression and four showed clinical or radiological disease recurrences at B-cell reconstitution. Mild-to-moderate infusion related adverse events occurred in two patients. Leukopenia developed in seven patients and serum immunoglobulin suppression in five patients with no significant age effect on the risk of their development. None developed severe life-threatening events. Rituximab-induced B-cell suppression was associated with absence of relapses in 10 patients who were MOG-positive with recurrent disease. Rituximab was well tolerated. The most frequent adverse effects were hypogammaglobulinemia and leukopenia. We recommend monitoring of complete blood counts and immunoglobulins in this population.Myelin oligodendrocyte glycoprotein (MOG) antibodies are seen in the serum of 25% to 58% of children who present with a first acquired demyelinating syndrome, 1-5 and may also be seen in adults presenting with acute neuroinflammation (4-15%). 3,5-7 Twenty-five per cent to almost 75% will continue to have high titers of MOG antibodies after presenting acutely, 8 with considerable risk of relapse. 9 In one adult-based cohort of 252 patients in the UK with elevated MOG antibodies, 36% had relapses within a median duration of 16 months. 8 Steroids, intravenous immunoglobulin (IVIG), and plasma exchange 10 may be beneficial in reducing symptomatology at the time of an acute event, but are unlikely to decrease the risk of relapse.There is variable support in the literature for the relevance of persistent MOG antibody status for predicting recurrent disease. 11,12 However, prevention of recurrence in the face of repeated neuroinflammatory events prevent future disability. Given the potential for irreversible neurological disability, 13 and the robust response of other antibody-mediated conditions to B-cell suppression with rituximab therapy, 14 we used a standardized treatment protocol using rituximab in children with recurrent central nervous system (CNS) inflammatory disease and persistent MOG antibody positivity 12 weeks or more af...

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“…Olgularımızda yedi hastada Anti-MOG antikoru [ADEM(1), MDEM(1), NMOSD(2), KIS(1), ON(2)] pozitif bulunmuştur. MOG antikoru ile ilişkili NMO/NMOSD hastalarının başlangıç yaşı daha küçüktür (17) . Bu çalışmada sunulan NMOSD hastalarının da başlangıç yaşları 3 ve 6 olup, literatür ile uyumlu bulunmuştur.…”

Section: Discussionunclassified

“…Anti-MOG antikor pozitif olguların çoğunluğu monofazik seyirli olduğu bildirilmesine karşın, son zamanlarda yapılan çalışmalar, anti-MOG antikor ilişkili demiyelinizan hastalıkların relapslar ile seyredebileceğini göstermiştir (15,17,18) . Bizim de üç olgumuzda relaps izlendi.…”

Section: Discussionunclassified

Acquired Demyelinating Syndrome: Single Center Experience

Serin¹,

Şimşek²,

Kanmaz³

et al. 2019

BUCH

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Bu hastalardan birinde hem anti-aquaporin 4 hem de anti-miyelin oligodendrosit antikorları pozitif bulundu. Nörogörüntülemede serebral beyaz cevher, spinal kord ve optik sinir tutulumuyla uyumlu bulgular mevcuttu. Tüm hastalara atak sırasında yüksek doz metilprednizolon tedavisi başlandı ve idame oral steroid ile devam edildi. Relapsları olan hastalarda uzun dönem tedavide azatioprin ve aylık IVIG tedavileri uygulandı. Sonuç: Çocukluk çağı edinsel demiyelinizan hastalıkları multiple sklerozun ilk atağı olabileceğinden hastaların takibi ve ayırıcı tanı için başvuruda ayrıntılı serolojik testlerin, BOS incelemelerinin yapılması önemlidir.

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Retrospective analysis of children with myelin oligodendrocyte glycoprotein antibody-related disorders

Cited by 35 publications

References 28 publications

Diagnostic Considerations in Acute Disseminated Encephalomyelitis and the Interface with MOG Antibody

Diagnostic Considerations in Acute Disseminated Encephalomyelitis and the Interface with MOG Antibody

Rituximab in children with myelin oligodendrocyte glycoprotein antibody and relapsing neuroinflammatory disease

Acquired Demyelinating Syndrome: Single Center Experience

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