Long-term remission with rituximab in refractory leucine-rich glioma inactivated 1 antibody encephalitis

Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic...

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Veröffentlicht in:Journal of neuroimmunology Jg. 271; H. 1-2; S. 66 - 68
Hauptverfasser: Brown, J. William L., Martin, Peter J., Thorpe, John W., Michell, Andrew W., Coles, Alasdair J., Cox, Amanda L., Vincent, Angela, Zandi, Michael S.
Format: Journal Article
Sprache:Englisch
Veröffentlicht: Netherlands Elsevier B.V 15.06.2014
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ISSN:0165-5728, 1872-8421, 1872-8421
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Abstract Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic seizures (FBDS) and received rituximab after relapsing or refractory disease. Both cases achieved sustained clinical remission of up to 15 and 56months respectively. Rituximab use allowed withdrawal of corticosteroids and was well tolerated. Randomized clinical trials are needed in LGI1 encephalitis and other autoimmune encephalitides.
AbstractList Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic seizures (FBDS) and received rituximab after relapsing or refractory disease. Both cases achieved sustained clinical remission of up to 15 and 56 months respectively. Rituximab use allowed withdrawal of corticosteroids and was well tolerated. Randomized clinical trials are needed in LGI1 encephalitis and other autoimmune encephalitides.Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic seizures (FBDS) and received rituximab after relapsing or refractory disease. Both cases achieved sustained clinical remission of up to 15 and 56 months respectively. Rituximab use allowed withdrawal of corticosteroids and was well tolerated. Randomized clinical trials are needed in LGI1 encephalitis and other autoimmune encephalitides.
Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic seizures (FBDS) and received rituximab after relapsing or refractory disease. Both cases achieved sustained clinical remission of up to 15 and 56 months respectively. Rituximab use allowed withdrawal of corticosteroids and was well tolerated. Randomized clinical trials are needed in LGI1 encephalitis and other autoimmune encephalitides.
Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic seizures (FBDS) and received rituximab after relapsing or refractory disease. Both cases achieved sustained clinical remission of up to 15 and 56months respectively. Rituximab use allowed withdrawal of corticosteroids and was well tolerated. Randomized clinical trials are needed in LGI1 encephalitis and other autoimmune encephalitides.
Abstract Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on the treatment of relapsing or refractory cases and long-term outcomes. Two case reports are presented. Both cases had faciobrachial dystonic seizures (FBDS) and received rituximab after relapsing or refractory disease. Both cases achieved sustained clinical remission of up to 15 and 56 months respectively. Rituximab use allowed withdrawal of corticosteroids and was well tolerated. Randomized clinical trials are needed in LGI1 encephalitis and other autoimmune encephalitides.
Author Coles, Alasdair J.
Cox, Amanda L.
Brown, J. William L.
Martin, Peter J.
Vincent, Angela
Thorpe, John W.
Zandi, Michael S.
Michell, Andrew W.
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  givenname: J. William L.
  surname: Brown
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  email: william.brown@doctors.org.uk
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
– sequence: 2
  givenname: Peter J.
  surname: Martin
  fullname: Martin, Peter J.
  email: peter.martin@addenbrookes.nhs.uk
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
– sequence: 3
  givenname: John W.
  surname: Thorpe
  fullname: Thorpe, John W.
  email: john.thorpe@addenbrookes.nhs.uk
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
– sequence: 4
  givenname: Andrew W.
  surname: Michell
  fullname: Michell, Andrew W.
  email: andrew.michell@addenbrookes.nhs.uk
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
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  givenname: Alasdair J.
  surname: Coles
  fullname: Coles, Alasdair J.
  email: ajc1020@medschl.cam.ac.uk
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
– sequence: 6
  givenname: Amanda L.
  surname: Cox
  fullname: Cox, Amanda L.
  email: amandacox1@nhs.net
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
– sequence: 7
  givenname: Angela
  surname: Vincent
  fullname: Vincent, Angela
  email: angela.vincent@ndcn.ox.ac.uk
  organization: Neurosciences Group, Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, Oxford, United Kingdom
– sequence: 8
  givenname: Michael S.
  orcidid: 0000-0002-9612-9401
  surname: Zandi
  fullname: Zandi, Michael S.
  email: msz20@cam.ac.uk
  organization: Department of Clinical Neurosciences, Addenbrooke's Hospital, University of Cambridge, United Kingdom
BackLink https://www.ncbi.nlm.nih.gov/pubmed/24703099$$D View this record in MEDLINE/PubMed
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Issue 1-2
Keywords LGI1
Autoantibody
Rituximab
VGKC
Encephalitis
Cognition
Language English
License Copyright © 2014 Elsevier B.V. All rights reserved.
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  ident: 10.1016/j.jneuroim.2014.03.012_bb0060
  article-title: Defining and treating leucine-rich glioma inactivated 1 antibody associated autoimmunity
  publication-title: Brain
  doi: 10.1093/brain/awt256
– volume: 350
  start-page: 2572
  year: 2004
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  article-title: Efficacy of B-cell-targeted therapy with rituximab in patients with rheumatoid arthritis
  publication-title: N. Engl. J. Med.
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Snippet Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed reports on...
Abstract Autoimmune encephalitis associated with antibodies to leucine-rich glioma inactivated 1 (LGI1) is recently described and there is a lack of detailed...
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StartPage 66
SubjectTerms Adult
Aged
Allergy and Immunology
Antibodies - blood
Antibodies, Monoclonal, Murine-Derived - therapeutic use
Autoantibody
Cognition
Encephalitis
Encephalitis - blood
Encephalitis - drug therapy
Encephalitis - immunology
Female
Humans
Immunologic Factors - therapeutic use
LGI1
Male
Neurology
Proteins - immunology
Rituximab
Secondary Prevention
Seizures - drug therapy
Treatment Outcome
VGKC
Title Long-term remission with rituximab in refractory leucine-rich glioma inactivated 1 antibody encephalitis
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https://dx.doi.org/10.1016/j.jneuroim.2014.03.012
https://www.ncbi.nlm.nih.gov/pubmed/24703099
https://www.proquest.com/docview/1526127464
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