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March 2019; 6 (2) ArticleOpen Access

Cranial nerve involvement in patients with MOG antibody–associated disease

Alvaro Cobo-Calvo, Xavier Ayrignac, Philippe Kerschen, Philippe Horellou, Francois Cotton, Pierre Labauge, Sandra Vukusic, Kumaran Deiva, Ché Serguera, Romain Marignier
First published February 1, 2019, DOI: https://doi.org/10.1212/NXI.0000000000000543
Alvaro Cobo-Calvo
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Xavier Ayrignac
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Philippe Kerschen
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Philippe Horellou
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Francois Cotton
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Pierre Labauge
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Sandra Vukusic
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Kumaran Deiva
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Ché Serguera
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Romain Marignier
From the Service de neurologie (A.C.-C., S.V., R.M.), sclérose en plaques, pathologies de la myéline et neuroinflammation and Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle (MIRCEM)– Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, France; Lyon Neuroscience Research Center (A.C.-C., R.M.), U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France; Service de sclérose en plaques (X.A., P.L.), Hôpital Universitaire de Montpellier, France; Service de Neurologie (P.K.), Centre hospitalier de Luxembourg; Inserm (P.H.), U 1184, Center for Immunology of Viral Infections and Autoimmune Diseases, Université Paris-Sud 11, CEA, DSV/iMETI, Division of Immuno-Virology, IDMIT, Faculté de médecine, Le Kremlin-Bicêtre Cedex, France; Service de Radiologie (F.C.), Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France; Université Claude Bernard Lyon 1 (F.C., S.V., R.M.), F-69100 Villeurbanne, France; Lyon's Neuroscience Research Center (S.V.), Observatoire Français de la Sclérose en Plaques, INSERM 1028 et CNRS UMR5292, Lyon, France; Service de neurologie pédiatrique (K.D.), Centre de référence pour les maladies inflammatoires rares du cerveau et de la moelle, Le Kremlin-Bicêtre, France; and INSERM US27 MIRCen (C.S.), CEA, Fontenay-aux-Roses, France.
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Citation
Cranial nerve involvement in patients with MOG antibody–associated disease
Alvaro Cobo-Calvo, Xavier Ayrignac, Philippe Kerschen, Philippe Horellou, Francois Cotton, Pierre Labauge, Sandra Vukusic, Kumaran Deiva, Ché Serguera, Romain Marignier
Neurol Neuroimmunol Neuroinflamm Mar 2019, 6 (2) e543; DOI: 10.1212/NXI.0000000000000543

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Abstract

Objective To describe clinical and radiologic features of cranial nerve (CN) involvement in patients with myelin oligodendrocyte glycoprotein antibodies (MOG-IgG) and to assess the potential underlying mechanism of CN involvement using a nonhuman primate (NHP) model.

Methods Epidemiologic, clinical, and radiologic features from a national cohort of 273 MOG-IgG–positive patients were retrospectively reviewed for CN involvement between January 2014 and January 2018. MOG-IgG binding was evaluated in CNS, CN, and peripheral nerve tissues from NHP.

Results We identified 3 MOG-IgG–positive patients with radiologic and/or clinical CN involvement. Two patients displayed either trigeminal or vestibulocochlear nerve lesions at the root level, and the remaining patient had an oculomotor nerve involvement at the root exit and at the cisternal level. Additional CNS involvement was found in all 3 patients. None of the 3 patients' sera recognized MOG expression in CN of NHP.

Conclusion Craneal nerve involvement can coexist in patients with MOG antibody disease, although the underlying pathophysiology remains elusive.

Glossary

AQP4=
aquaporin-4;
CN=
cranial nerve;
hMOG=
human MOG;
MBP=
myelin basic protein;
MFI=
mean fluorescence intensity;
MOG=
myelin oligodendrocyte glycoprotein;
MTP=
methylprednisolone;
NHP=
nonhuman primate;
NMOSD=
neuromyelitis optica spectrum disorder;
OCB=
oligoclonal band;
OFSEP=
Observatoire Français de la Sclérose en Plaques;
RT=
room temperature

Footnotes

  • ↵* These authors contributed equally to the manuscript.

  • Funding information and disclosures are provided at the end of the article. Full disclosure form information provided by the authors is available with the full text of this article at Neurology.org/NN.

  • The Article Processing Charge was funded by the authors.

  • Received October 8, 2018.
  • Accepted in final form December 7, 2018.
  • Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology.

This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND), which permits downloading and sharing the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.

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