Glutamate receptor δ2 serum antibodies in pediatric opsoclonus myoclonus ataxia syndrome

Berridge, G.; Menassa, David A.; Moloney, Teresa C.; Waters, Patrick; Welding, Imogen; Thomsen, Selina; Zuberi, Sameer M.; Fischer, Román; Aricescu, A. Radu; Pike, Michael; Dale, Russell C.; Kessler, Benedikt M.; Vincent, Angela; Lim, Ming; Irani, Sarosh R.; Lang, Bethan

doi:10.1212/wnl.0000000000006035

Cited by 49 publications

(22 citation statements)

References 30 publications

(42 reference statements)

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“…Furthermore, there is a strong association between OMS and neuroblastoma [ 2 ]. Children with OMS display functionally active auto-antibodies, pro-inflammatory changes in the cytokine network and response to immunosuppressive treatment, thus suggesting underlying auto-immune phenomena [ 3 ].…”

Section: Introductionmentioning

confidence: 99%

Evolving Cognitive Dysfunction in Children with Neurologically Stable Opsoclonus–Myoclonus Syndrome

Goh

Scarff²,

Satariano

et al. 2020

Children

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Cognitive and acquired neurodevelopmental deficits have been reported in children with opsoclonus–myoclonus syndrome (OMS) and are known to be associated with more severe and relapsing disease course. However, there is a paucity of data regarding cognitive dysfunction in children with stable neurological disease. We report three children with OMS and evolving cognitive dysfunction in the context of a mild disease course. The children’s ages at disease onset were between 17 and 35 months and they were followed up for 4–10 years. Neuroblastoma was identified in one child. OMS severity scores ranged between 8 and 12/15 at presentation. They underwent immunotherapy and all were in remission by 7 months (range 4–13 months), with treatment maintained for 1 year. One child remained relapse-free, while two others had one clinical relapse each and were immunotherapy-responsive again. In all cases, evolving cognitive dysfunction was reported despite being in remission and stable off treatment for a median of 20 months (range of 12–31 months; two OMS scores of 0/15 and one of 2/15). In children with OMS who have completed treatment and have made full or near full neurological recovery, concerns remain regarding long-term outcome in terms of future learning and cognitive development.

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Section: Introductionmentioning

confidence: 99%

Evolving Cognitive Dysfunction in Children with Neurologically Stable Opsoclonus–Myoclonus Syndrome

Goh

Scarff²,

Satariano

et al. 2020

Children

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“…Occasionally, Ri-antibodies can be found and are highly suggestive of ovarian or breast cancer. Recently, OMS in the context of NMDAR, GABA(b), GlyR-antibodies, and more recently, yet not reproduced by other centers, glutamate receptor δ2 antibodies, have been described [ 44 , 45 ]. Importantly, these antibodies are not OMS-specific [ 1 ].…”

Section: The Clinical Approachmentioning

confidence: 99%

Antibody-related movement disorders – a comprehensive review of phenotype-autoantibody correlations and a guide to testing

Gövert

Leypoldt

Junker

et al. 2020

Neurol. Res. Pract.

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Background Over the past decade increasing scientific progress in the field of autoantibody–mediated neurological diseases was achieved. Movement disorders are a frequent and often prominent feature in such diseases which are potentially treatable. Main body Antibody-mediated movement disorders encompass a large clinical spectrum of diverse neurologic disorders occurring either in isolation or accompanying more complex autoimmune encephalopathic diseases. Since autoimmune movement disorders can easily be misdiagnosed as neurodegenerative or metabolic conditions, appropriate immunotherapy can be delayed or even missed. Recognition of typical clinical patterns is important to reach the correct diagnosis. Conclusion There is a growing number of newly discovered antibodies which can cause movement disorders. Several antibodies can cause distinctive phenotypes of movement disorders which are important to be aware of. Early diagnosis is important because immunotherapy can result in major improvement. In this review article we summarize the current knowledge of autoimmune movement disorders from a point of view focused on clinical syndromes. We discuss associated clinical phenomenology and antineuronal antibodies together with alternative etiologies with the aim of providing a diagnostic framework for clinicians considering underlying autoimmunity in patients with movement disorders.

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“…Slides were left to dry in the fume hood before proceeding with dehydration procedure by immersion in progressive concentration of ethanol solutions, followed by p-Xylene. 12 Slides were mounted with DPX mountant for histology (SigmaeAldrich, St. Louis, Mo). Sections were viewed with a light microscope (Nikon Eclipse E400; Nikon, Melville, NY), and images were taken with the Aperio ScanScope (Leica Biosystems, Wetzlar, Germany).…”

Section: Immunohistologymentioning

confidence: 99%

Searching for Serum Antibodies to Neuronal Proteins in Patients With Myalgic Encephalopathy/Chronic Fatigue Syndrome

Giannoccaro

Cossins

Sørland

et al. 2019

Clinical Therapeutics

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Purpose: A role for the immune system in causing myalgic encephalopathy/chronic fatigue syndrome (ME/CFS) is long suspected, but few studies have looked for specific autoantibodies that might contribute to the symptoms. Our aim was to look for evidence of antibodies to neuronal proteins in patients with ME/CSF.Methods: Sera samples from 50 patients and 50 healthy individuals were sent coded to the Neuroimmunology Laboratory in Oxford. Screening for antibody binding to neuronal tissue was performed on brain tissue and neuronal cultures. Specific serum antibodies were assessed by antigenspecific cell-based assays and radioimmunoassays. After antibody testing, the associations between seropositive status and clinical data were investigated.Findings: Overall, 8 patients and 11 participants were found to have some serum immunoreactivity toward neuronal or neuromuscular junction proteins, but only 1 patient and 2 participants had specific serum antibodies. Nevertheless, seropositive status in patients with ME was associated with shorter duration since onset and a more severe disease.Implications:The results indicate no overall increased frequency of antibodies to neuronal proteins in ME/CSF and no evidence of a specific antibody that might be causative or contribute to clinical features in patients. However, the association of seropositive status with shorter duration of disease and more severe symptoms suggests a possible role of antibodies at onset in some patients and should be the focus of future studies. (Clin Ther. 2019;41:836e847)

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Glutamate receptor δ2 serum antibodies in pediatric opsoclonus myoclonus ataxia syndrome

Cited by 49 publications

References 30 publications

Evolving Cognitive Dysfunction in Children with Neurologically Stable Opsoclonus–Myoclonus Syndrome

Evolving Cognitive Dysfunction in Children with Neurologically Stable Opsoclonus–Myoclonus Syndrome

Antibody-related movement disorders – a comprehensive review of phenotype-autoantibody correlations and a guide to testing

Searching for Serum Antibodies to Neuronal Proteins in Patients With Myalgic Encephalopathy/Chronic Fatigue Syndrome

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