N-methyl-D-aspartate receptor antibodies in post–herpes simplex virus encephalitis neurological relapse

Authors

  • Yael Hacohen MRCPCH,

    1. Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, United Kingdom
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  • Kumaran Deiva MD, PhD,

    1. Assistance Publique Hôpitaux de Paris, Hopitaux Universitaires Paris Sud, Pediatric Neurology Department and National Referral Center for Neuroinflammatory Diseases and Université Paris Sud, Le Kremlin-Bicêtre Hospital (BH), Paris, France
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  • Phillipa Pettingill BSc,

    1. Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, United Kingdom
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  • Patrick Waters PhD,

    1. Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, United Kingdom
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  • Ata Siddiqui MD, FRCR,

    1. Department of Neuroradiology, Kings College Hospital, King's Health Partners Academic Health Science Centre, London, United Kingdom
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  • Pascale Chretien MD, PhD,

    1. Assistance Publique Hôpitaux de Paris, Hopitaux Universitaires Paris Sud, Immunology Department, Université Paris Sud, Le Kremlin-Bicêtre Hospital (BH), Paris, France
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  • Esse Menson MRCPCH, PhD,

    1. Paediatric Infectious Diseases Department, Evelina Children's Hospital (ECH) at Guy's and St Thomas' National Health Service (NHS) Foundation Trust, King's Health Partners Academic Health Science Centre, London, United Kingdom
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  • Jean-Pierre Lin MRCP, PhD,

    1. Children's Neurosciences, Evelina Children's Hospital (ECH) at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre, London, United Kingdom
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  • Marc Tardieu MD, PhD,

    1. Assistance Publique Hôpitaux de Paris, Hopitaux Universitaires Paris Sud, Pediatric Neurology Department and National Referral Center for Neuroinflammatory Diseases and Université Paris Sud, Le Kremlin-Bicêtre Hospital (BH), Paris, France
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  • Angela Vincent FRS, FMedSci,

    Corresponding author
    1. Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, United Kingdom
    • Correspondence to: Dr. Ming Lim, Children's Neurosciences, Evelina Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Sciences Centre, London SE1 7EH, UK; ming.lim@gstt.nhs.uk; and Prof Angela Vincent, Level 6, West Wing, Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, OX3 9DU, UK angela.vincent@ndnc.ox.ac.uk

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  • Ming J. Lim MRCP, PhD

    Corresponding author
    1. Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, United Kingdom
    2. Children's Neurosciences, Evelina Children's Hospital (ECH) at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre, London, United Kingdom
    • Correspondence to: Dr. Ming Lim, Children's Neurosciences, Evelina Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Sciences Centre, London SE1 7EH, UK; ming.lim@gstt.nhs.uk; and Prof Angela Vincent, Level 6, West Wing, Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, OX3 9DU, UK angela.vincent@ndnc.ox.ac.uk

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  • Funding agencies: National Institute for Health Research (NIHR) Oxford Biomedical Research Centre, Oxford University Hospitals NHS Trust and the University of Oxford (to Y.H., P.W., and A.V.); NHS Specialized Services for Rare Diseases (neuromyelitis optica; to P.W. and A.V.).

  • Relevant conflicts of interest/financial disclosures: AV and the University of Oxford, hold patents and receive royalties and payments for antibody assays in neurologic diseases.

  • Full financial disclosures and author roles may be found in the online version of this article.

ABSTRACT

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. © 2013 International Parkinson and Movement Disorder Society

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