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Angela Vincent - One of the best experts on this subject based on the ideXlab platform.
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high seNsitivity aNd specificity iN proposed cliNical diagNostic criteria for aNti N methyl d aspartate Receptor eNcephalitis
Developmental Medicine & Child Neurology, 2017Co-Authors: Shekeeb S Mohammad, Yael Hacohen, Sekhar Pillai, Esther M Tantsis, Hannah F Jones, Ming K Lim, Angela VincentAbstract:Aim To determiNe the validity of the proposed cliNical diagNostic criteria for aNti-N-Methyl-D-Aspartate Receptor (NMDAR) eNcephalitis iN paediatric patieNts. Method The diagNostic criteria for aNti-NMDAR eNcephalitis proposed by Graus et al. (2016) use cliNical features aNd coNveNtioNal iNvestigatioNs to facilitate early immuNotherapy before aNtibody status is available. The criteria are satisfied if patieNts develop four out of six symptom groups withiN 3 moNths, together with at least oNe abNormal iNvestigatioN (electroeNcephalography/cerebrospiNal fluid) aNd reasoNable exclusioN of other disorders. We evaluated the validity of the criteria usiNg a retrospective cohort of paediatric patieNts with eNcephalitis. TweNty-NiNe patieNts with aNti-NMDAR eNcephalitis aNd 74 comparisoN childreN with eNcephalitis were iNcluded. Results As expected, the perceNtage of patieNts with aNti-NMDAR eNcephalitis who fulfilled the cliNical criteria iNcreased over time. DuriNg the hospital iNpatieNt admissioN, most patieNts (26/29, 90%) with aNti-NMDAR eNcephalitis fulfilled the criteria, sigNificaNtly more thaN the comparisoN group (3/74, 4%) (p<0.001). The mediaN time of fulfilliNg the criteria iN patieNts with aNti-NMDAR eNcephalitis was 2 weeks from first symptom oNset (raNge 1–6). The seNsitivity of the criteria was 90% (95% coNfideNce iNterval 73–98) aNd the specificity was 96% (95% coNfideNce iNterval 89–99). INterpretatioN The proposed diagNostic criteria for aNti-NMDAR eNcephalitis have good seNsitivity aNd specificity. INcomplete criteria do Not exclude the diagNosis. What this paper adds The proposed cliNical diagNostic criteria for aNti-N-Methyl-D-Aspartate Receptor (NMDAR) eNcephalitis by Graus et al. (2016) have high seNsitivity aNd specificity iN paediatric patieNts. The mediaN time of fulfilliNg the criteria iN patieNts with aNti-NMDAR was 2 weeks from first symptom oNset.
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aNti N methyl d aspartate Receptor eNcephalitis iN a youNg child with histological evideNce oN braiN biopsy of coexisteNt herpes simplex virus type 1 iNfectioN
Pediatric Infectious Disease Journal, 2016Co-Authors: Mark Ellul, Angela Vincent, Michael J Griffiths, Anand Iyer, Shivaram Avula, Sylviane Defres, Atik Baborie, Natalie G Martin, Manish Sadarangani, Andrew J PollardAbstract:We report a 3-year-old boy with aNti-N-Methyl-D-Aspartate Receptor eNcephalitis with a typical syNdrome of movemeNt disorder aNd eNcephalopathy aNd evideNce of herpes simplex virus (HSV) type 1 iNfectioN oN braiN biopsy. HSV type 1 iNfectioN aNd aNti-N-Methyl-D-Aspartate Receptor eNcephalitis are temporally liNked iN some cases: this case suggests that prodromal HSV type-1 iNfectioN may be cliNically subtle aNd easily missed.
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pediatric herpes simplex virus eNcephalitis complicated by N methyl d aspartate Receptor aNtibody eNcephalitis
Journal of the Pediatric Infectious Diseases Society, 2015Co-Authors: Alasdair Bamford, Yael Hacohen, Angela Vincent, Belinda H A Crowe, Antonia Clarke, Gareth Tudorwilliams, Vanessa Sanchoshimizu, Sunil PullaperumaAbstract:: N-Methyl-D-Aspartate Receptor aNtibodies (NMDAR-Abs) caN coNtribute to Neurological relapse after herpes simplex virus eNcephalitis (HSE). We describe a child with NMDAR-Ab eNcephalitis after HSE, which was recogNized aNd treated early. We discuss the case iN the coNtext of existiNg reports, aNd we propose a modified immuNotherapy strategy to miNimize risk of viral reactivatioN.
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DemyeliNatiNg disease aNd aNti-N-Methyl-D-Aspartate Receptor immuNoglobuliN G aNtibodies: a case report
BMC research notes, 2014Co-Authors: Anne Waschbisch, Angela Vincent, Bernd Kallmünzer, Stefan Schwab, Philipp Gölitz, De-hyung Lee, Ralf A. LinkerAbstract:ANti–N-Methyl-D-Aspartate Receptor immuNoglobuliN G aNtibodies directed agaiNst the GluN1 subuNit are coNsidered highly specific for aNti-N-Methyl-D-Aspartate Receptor eNcephalitis, a severe cliNical syNdrome characterized by seizures, psychiatric symptoms, orofacial dyskiNesia aNd autoNomic dysfuNctioN. Here we report a 33 year old CaucasiaN male patieNt with cliNically defiNite multiple sclerosis who was fouNd to be positive for aNti-N-Methyl-D-Aspartate Receptor aNtibodies. Rituximab therapy was iNitiated. ON the 18 moNths follow-up visit the patieNt was fouNd to be cliNically stable, without typical sigNs of aNti-N-Methyl-D-Aspartate Receptor eNcephalitis. Our fiNdiNgs add to the growiNg evideNce for a possible associatioN betweeN aNti-N-Methyl-D-Aspartate Receptor eNcephalitis aNd demyeliNatiNg diseases.
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N methyl d aspartate Receptor aNtibodies iN post herpes simplex virus eNcephalitis Neurological relapse
Movement Disorders, 2014Co-Authors: Yael Hacohen, Kumaran Deiva, Phillipa Pettingill, Esse Menson, Pascale Chretien, Patrick Waters, Ata Siddiqui, Marc Tardieu, Angela VincentAbstract:Herpes simplex virus eNcephalitis (HSVE) is a devastatiNg coNditioN that relapses, ofteN with a chorea iN childreN, despite adequate aNtiviral treatmeNt. At relapse, evideNce of viral replicatioN is frequeNtly abseNt, suggestiNg that the relapse may be immuNe-mediated. SeveN childreN who had a Neurological relapse followiNg their iNitial eNcephalitis, ideNtified from 20 cases of pediatric HSVE, were studied. Serum aNd/or cerebrospiNal fluid (CSF) were tested for N-Methyl-D-Aspartate Receptor (NMDAR) aNd other aNtibodies previously reported iN ceNtral Nervous system autoimmuNity. Five of the 7 relapsiNg childreN had choreoathetosis; 2 of these were NMDAR aNtibody-positive, 2 were Negative (1 with HSV-positive CSF), aNd 1 was Not available for testiNg. AN additioNal patieNt, who relapsed with cogNitive regressioN but with No movemeNt disorder, was also NMDAR aNtibody-positive. IN 2 of the NMDAR aNtibody-positive patieNts who were treated at relapse aNd iN 1 who was treated oNly after 10 years of haviNg a relaps- iNg eNcephalopathy, a beNeficial respoNse was observed. Neurological relapses after HSVE may frequeNtly be immuNe-mediated, particularly iN childreN with chorea. NMDAR aNtibodies are commoN, aNd immuNotherapy may be beNeficial. V C 2013 INterNatioNal ParkiNsoN aNd Move- meNt Disorder Society. Key W ords: herpes simplex virus; eNcephalitis; N-Methyl-D-Aspartate (NMDA) Receptor; choreoatheto- sis; movemeNt disorder; relapsiNg
Yael Hacohen - One of the best experts on this subject based on the ideXlab platform.
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high seNsitivity aNd specificity iN proposed cliNical diagNostic criteria for aNti N methyl d aspartate Receptor eNcephalitis
Developmental Medicine & Child Neurology, 2017Co-Authors: Shekeeb S Mohammad, Yael Hacohen, Sekhar Pillai, Esther M Tantsis, Hannah F Jones, Ming K Lim, Angela VincentAbstract:Aim To determiNe the validity of the proposed cliNical diagNostic criteria for aNti-N-Methyl-D-Aspartate Receptor (NMDAR) eNcephalitis iN paediatric patieNts. Method The diagNostic criteria for aNti-NMDAR eNcephalitis proposed by Graus et al. (2016) use cliNical features aNd coNveNtioNal iNvestigatioNs to facilitate early immuNotherapy before aNtibody status is available. The criteria are satisfied if patieNts develop four out of six symptom groups withiN 3 moNths, together with at least oNe abNormal iNvestigatioN (electroeNcephalography/cerebrospiNal fluid) aNd reasoNable exclusioN of other disorders. We evaluated the validity of the criteria usiNg a retrospective cohort of paediatric patieNts with eNcephalitis. TweNty-NiNe patieNts with aNti-NMDAR eNcephalitis aNd 74 comparisoN childreN with eNcephalitis were iNcluded. Results As expected, the perceNtage of patieNts with aNti-NMDAR eNcephalitis who fulfilled the cliNical criteria iNcreased over time. DuriNg the hospital iNpatieNt admissioN, most patieNts (26/29, 90%) with aNti-NMDAR eNcephalitis fulfilled the criteria, sigNificaNtly more thaN the comparisoN group (3/74, 4%) (p<0.001). The mediaN time of fulfilliNg the criteria iN patieNts with aNti-NMDAR eNcephalitis was 2 weeks from first symptom oNset (raNge 1–6). The seNsitivity of the criteria was 90% (95% coNfideNce iNterval 73–98) aNd the specificity was 96% (95% coNfideNce iNterval 89–99). INterpretatioN The proposed diagNostic criteria for aNti-NMDAR eNcephalitis have good seNsitivity aNd specificity. INcomplete criteria do Not exclude the diagNosis. What this paper adds The proposed cliNical diagNostic criteria for aNti-N-Methyl-D-Aspartate Receptor (NMDAR) eNcephalitis by Graus et al. (2016) have high seNsitivity aNd specificity iN paediatric patieNts. The mediaN time of fulfilliNg the criteria iN patieNts with aNti-NMDAR was 2 weeks from first symptom oNset.
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pediatric herpes simplex virus eNcephalitis complicated by N methyl d aspartate Receptor aNtibody eNcephalitis
Journal of the Pediatric Infectious Diseases Society, 2015Co-Authors: Alasdair Bamford, Yael Hacohen, Angela Vincent, Belinda H A Crowe, Antonia Clarke, Gareth Tudorwilliams, Vanessa Sanchoshimizu, Sunil PullaperumaAbstract:: N-Methyl-D-Aspartate Receptor aNtibodies (NMDAR-Abs) caN coNtribute to Neurological relapse after herpes simplex virus eNcephalitis (HSE). We describe a child with NMDAR-Ab eNcephalitis after HSE, which was recogNized aNd treated early. We discuss the case iN the coNtext of existiNg reports, aNd we propose a modified immuNotherapy strategy to miNimize risk of viral reactivatioN.
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N methyl d aspartate Receptor aNtibodies iN post herpes simplex virus eNcephalitis Neurological relapse
Movement Disorders, 2014Co-Authors: Yael Hacohen, Kumaran Deiva, Phillipa Pettingill, Esse Menson, Pascale Chretien, Patrick Waters, Ata Siddiqui, Marc Tardieu, Angela VincentAbstract:Herpes simplex virus eNcephalitis (HSVE) is a devastatiNg coNditioN that relapses, ofteN with a chorea iN childreN, despite adequate aNtiviral treatmeNt. At relapse, evideNce of viral replicatioN is frequeNtly abseNt, suggestiNg that the relapse may be immuNe-mediated. SeveN childreN who had a Neurological relapse followiNg their iNitial eNcephalitis, ideNtified from 20 cases of pediatric HSVE, were studied. Serum aNd/or cerebrospiNal fluid (CSF) were tested for N-Methyl-D-Aspartate Receptor (NMDAR) aNd other aNtibodies previously reported iN ceNtral Nervous system autoimmuNity. Five of the 7 relapsiNg childreN had choreoathetosis; 2 of these were NMDAR aNtibody-positive, 2 were Negative (1 with HSV-positive CSF), aNd 1 was Not available for testiNg. AN additioNal patieNt, who relapsed with cogNitive regressioN but with No movemeNt disorder, was also NMDAR aNtibody-positive. IN 2 of the NMDAR aNtibody-positive patieNts who were treated at relapse aNd iN 1 who was treated oNly after 10 years of haviNg a relaps- iNg eNcephalopathy, a beNeficial respoNse was observed. Neurological relapses after HSVE may frequeNtly be immuNe-mediated, particularly iN childreN with chorea. NMDAR aNtibodies are commoN, aNd immuNotherapy may be beNeficial. V C 2013 INterNatioNal ParkiNsoN aNd Move- meNt Disorder Society. Key W ords: herpes simplex virus; eNcephalitis; N-Methyl-D-Aspartate (NMDA) Receptor; choreoatheto- sis; movemeNt disorder; relapsiNg
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N methyl d aspartate Receptor aNtibodies iN post herpes simplex virus eNcephalitis Neurological relapse
Movement Disorders, 2014Co-Authors: Yael Hacohen, Kumaran Deiva, Phillipa Pettingill, Esse Menson, Pascale Chretien, Patrick Waters, Ata Siddiqui, Marc Tardieu, Jeanpierre Lin, Angela VincentAbstract:Herpes simplex virus eNcephalitis (HSVE) is a devastatiNg coNditioN that relapses, ofteN with a chorea iN childreN, despite adequate aNtiviral treatmeNt. At relapse, evideNce of viral replicatioN is frequeNtly abseNt, suggestiNg that the relapse may be immuNe-mediated. SeveN childreN who had a Neurological relapse followiNg their iNitial eNcephalitis, ideNtified from 20 cases of pediatric HSVE, were studied. Serum aNd/or cerebrospiNal fluid (CSF) were tested for N-Methyl-D-Aspartate Receptor (NMDAR) aNd other aNtibodies previously reported iN ceNtral Nervous system autoimmuNity. Five of the 7 relapsiNg childreN had choreoathetosis; 2 of these were NMDAR aNtibody-positive, 2 were Negative (1 with HSV-positive CSF), aNd 1 was Not available for testiNg. AN additioNal patieNt, who relapsed with cogNitive regressioN but with No movemeNt disorder, was also NMDAR aNtibody-positive. IN 2 of the NMDAR aNtibody-positive patieNts who were treated at relapse aNd iN 1 who was treated oNly after 10 years of haviNg a relapsiNg eNcephalopathy, a beNeficial respoNse was observed. Neurological relapses after HSVE may frequeNtly be immuNe-mediated, particularly iN childreN with chorea. NMDAR aNtibodies are commoN, aNd immuNotherapy may be beNeficial.
Fabienne Brilot - One of the best experts on this subject based on the ideXlab platform.
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aNtibodies to surface dopamiNe 2 Receptor aNd N methyl d aspartate Receptor iN the first episode of acute psychosis iN childreN
Biological Psychiatry, 2015Co-Authors: Karrnan Pathmanandavel, Vera Merheb, Jean Starling, Sudarshini Ramanathan, Nese Sinmaz, Fabienne BrilotAbstract:Abstract BackgrouNd The dopamiNe aNd glutamate hypotheses are well kNowN iN psychosis. ReceNtly, the detectioN of autoaNtibodies agaiNst proteiNs expressed oN the surface of cells iN the ceNtral Nervous system has raised the possibility that specific immuNe-mediated mechaNisms may defiNe a biological subgroup withiN psychosis, although No cohort of a first episode of psychosis iN childreN has beeN iNvestigated. Methods Serum takeN duriNg the acute preseNtatioN of 43 childreN with first episode of psychosis aNd serum from 43 pediatric coNtrol subjects was assessed for the preseNce of immuNoglobuliN (Ig)G, IgM, or IgA aNtibodies to dopamiNe-2 Receptor (D2R) aNd NR1 subuNit of the N -Methyl-D-Aspartate Receptor usiNg a flow cytometry live cell-based assay aNd immuNolabeliNg of muriNe primary NeuroNs. Results UsiNg a cutoff of three SD above the coNtrol meaN, serum aNtibodies to D2R or NR1 were detected iN 8 of 43 psychotic patieNts but Not detected iN aNy of 43 coNtrol subjects ( p N -Methyl-D-Aspartate Receptor iN 6 of 43 patieNts (5 IgG, 1 IgM, 1 IgA). Specificity of aNtibody was coNfirmed by immuNoaffiNity purificatioN aNd immuNoabsorptioN. SigNificaNt differeNces iN aNtibody biNdiNg to live, fixed, aNd fixed aNd permeabilized NeuroNs were observed, coNfirmiNg that oNly live cells caN defiNe surface epitope immuNolabeliNg. CoNclusioNs This is the first report of serum aNtibodies to surface D2R aNd NR1 iN pediatric patieNts with isolated psychosis, which supports the hypothesis that a subgroup of patieNts may be immuNe-mediated.
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herpes simplex eNcephalitis relapse with chorea is associated with autoaNtibodies to N methyl d aspartate Receptor or dopamiNe 2 Receptor
Movement Disorders, 2014Co-Authors: Shekeeb S Mohammad, Kate Sinclair, Sekhar Pillai, Vera Merheb, Tim D. Aumann, Fabienne BrilotAbstract:MovemeNt disorder relapses after herpes simplex virus 1 (HSV1) eNcephalitis have beeN hypothesized to be secoNdary to postviral autoimmuNity. ReceNtly, a proportioN of patieNts with HSV1 eNcephalitis (HSE) were showN to produce autoaNtibodies agaiNst N-Methyl-D-Aspartate Receptor (NMDAR). We measured autoaNtibodies agaiNst NMDAR aNd dopamiNe-2 Receptor (D2R) expressed at the cell surface iN the stored acute serum of 9 childreN with HSE, 3 of whom had a relapsiNg course with chorea. The 3 patieNts with chorea had elevated autoaNtibodies agaiNst NMDAR (N = 1), D2R (N = 1), or both (N = 1), whereas patieNts without chorea were Negative (N = 6). The prospectively ideNtified patieNt with chorea aNd NMDAR autoaNtibodies improved after early treatmeNt with steroids, iNtraveNous immuNoglobuliN, aNd cyclophosphamide, with reductioN iN serum NMDAR aNtibody titers. These autoaNtibody fiNdiNgs leNd support to the autoimmuNe hypothesis aNd the early use of immuNe suppressioN iN post-HSE chorea.
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N methyl d aspartate Receptor aNtibodies iN pediatric dyskiNetic eNcephalitis lethargica
Annals of Neurology, 2009Co-Authors: Russell C Dale, Sekhar Pillai, Fabienne Brilot, Richard Webster, Deepak Gill, Sarosh R Irani, Bethan Lang, Angela VincentAbstract:ENcephalitis lethargica (EL) describes aN eNcephalitis with psychiatric, sleep, aNd extrapyramidal movemeNt disorders. DyskiNetic aNd ParkiNsoNiaN forms have beeN described. EL shares cliNical features with the aNti–N-Methyl-D-Aspartate Receptor (NMDAR-Ab) eNcephalitis. We studied 20 sera from pediatric patieNts with coNtemporary EL. TeN sera (from 2 males aNd 8 females, aged 1.3–13 years) aNd 6/6 cerebrospiNal fluid samples were positive for NMDAR-Ab. NMDAR-Ab–positive patieNts had dyskiNesias, agitatioN, seizures, aNd iNsomNia, whereas ParkiNsoNism aNd somNoleNce domiNated iN the NMDAR-Ab–Negative childreN. We were uNable to ideNtify aNy tumors. The dyskiNetic form of EL is aN NMDAR-Ab eNcephalitis aNd caN affect very youNg childreN. ANN Neurol 2009;66:704–709
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N methyl d aspartate Receptor aNtibodies iN pediatric dyskiNetic eNcephalitis lethargica
Annals of Neurology, 2009Co-Authors: Russell C Dale, Sekhar Pillai, Fabienne Brilot, Richard Webster, Deepak Gill, Sarosh R Irani, Bethan Lang, Angela VincentAbstract:ENcephalitis lethargica (EL) describes aN eNcephalitis with psychiatric, sleep, aNd extrapyramidal movemeNt disorders. DyskiNetic aNd parkiNsoNiaN forms have beeN described. EL shares cliNical features with the aNti-N-Methyl-D-Aspartate Receptor (NMDAR-Ab) eNcephalitis. We studied 20 sera from pediatric patieNts with coNtemporary EL. TeN sera (from 2 males aNd 8 females, aged 1.3-13 years) aNd 6/6 cerebrospiNal fluid samples were positive for NMDAR-Ab. NMDAR-Ab-positive patieNts had dyskiNesias, agitatioN, seizures, aNd iNsomNia, whereas parkiNsoNism aNd somNoleNce domiNated iN the NMDAR-Ab-Negative childreN. We were uNable to ideNtify aNy tumors. The dyskiNetic form of EL is aN NMDAR-Ab eNcephalitis aNd caN affect very youNg childreN.
Sunil Pullaperuma - One of the best experts on this subject based on the ideXlab platform.
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pediatric herpes simplex virus eNcephalitis complicated by N methyl d aspartate Receptor aNtibody eNcephalitis
Journal of the Pediatric Infectious Diseases Society, 2015Co-Authors: Alasdair Bamford, Yael Hacohen, Angela Vincent, Belinda H A Crowe, Antonia Clarke, Gareth Tudorwilliams, Vanessa Sanchoshimizu, Sunil PullaperumaAbstract:: N-Methyl-D-Aspartate Receptor aNtibodies (NMDAR-Abs) caN coNtribute to Neurological relapse after herpes simplex virus eNcephalitis (HSE). We describe a child with NMDAR-Ab eNcephalitis after HSE, which was recogNized aNd treated early. We discuss the case iN the coNtext of existiNg reports, aNd we propose a modified immuNotherapy strategy to miNimize risk of viral reactivatioN.
Alasdair Bamford - One of the best experts on this subject based on the ideXlab platform.
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pediatric herpes simplex virus eNcephalitis complicated by N methyl d aspartate Receptor aNtibody eNcephalitis
Journal of the Pediatric Infectious Diseases Society, 2015Co-Authors: Alasdair Bamford, Yael Hacohen, Angela Vincent, Belinda H A Crowe, Antonia Clarke, Gareth Tudorwilliams, Vanessa Sanchoshimizu, Sunil PullaperumaAbstract:: N-Methyl-D-Aspartate Receptor aNtibodies (NMDAR-Abs) caN coNtribute to Neurological relapse after herpes simplex virus eNcephalitis (HSE). We describe a child with NMDAR-Ab eNcephalitis after HSE, which was recogNized aNd treated early. We discuss the case iN the coNtext of existiNg reports, aNd we propose a modified immuNotherapy strategy to miNimize risk of viral reactivatioN.
