PittsburghV1, France, Analysis, bibRecord, 000311

Utility and safety of rituximab in pediatric autoimmune and inflammatory CNS disease

Identifieur interne : 000311 ( France/Analysis ); précédent : 000310; suivant : 000312

Utility and safety of rituximab in pediatric autoimmune and inflammatory CNS disease

Auteurs : Russell C. Dale [Australie] ; Fabienne Brilot [Australie] ; Lisa V. Duffy [Canada] ; Marinka Twilt [Canada] ; Amy T. Waldman [États-Unis] ; Sona Narula [États-Unis] ; Eyal Muscal [États-Unis] ; Kumaran Deiva [France] ; Erik Andersen [Australie] ; Michael R. Eyre [Royaume-Uni] ; Despina Eleftheriou [Royaume-Uni] ; Paul A. Brogan [Royaume-Uni] ; Rachel Kneen [Royaume-Uni] ; Gulay Alper [États-Unis] ; Banu Anlar [Turquie] ; Evangeline Wassmer [États-Unis] ; Kirsten Heineman [Royaume-Uni] ; Cheryl Hemingway [Royaume-Uni] ; Catherine J. Riney [Australie] ; Andrew Kornberg [Australie] ; Marc Tardieu [France] ; Amber Stocco [États-Unis] ; Brenda Banwell [États-Unis] ; Mark P. Gorman [Canada] ; Susanne M. Benseler [Canada] ; MING LIM [Royaume-Uni]

Source :

Neurology [ 0028-3878 ] ; 2014.

RBID : Pascal:14-0183455

Descripteurs français

Pascal (Inist)
- Pathologie du système nerveux, Sécurité, Rituximab, Enfant, Pédiatrie, Maladie autoimmune, Maladie inflammatoire, Système nerveux central.
Wicri :
- topic : Enfant, Pédiatrie.

English descriptors

KwdEn :
- Autoimmune disease, Central nervous system, Child, Inflammatory disease, Nervous system diseases, Pediatrics, Rituximab, Safety.

Abstract

Objective: To assess the utility and safety of rituximab in pediatric autoimmune and inflammatory disorders of the CNS. Methods: Multicenter retrospective study. Results: A total of 144 children and adolescents (median age 8 years, range 0.7-17; 103 female) with NMDA receptor (NMDAR) encephalitis (n = 39), opsoclonus myoclonus ataxia syndrome (n = 32), neuromyelitis optica spectrum disorders (n = 20), neuropsychiatric systemic lupus erythematosus (n = 18), and other neuroinflammatory disorders (n = 35) were studied. Rituximab was given after a median duration of disease of 0.5 years (range 0.05-9.5 years). Infusion adverse events were recorded in 18/144 (12.5%), including grade 4 (anaphylaxis) in 3. Eleven patients (7.6%) had an infectious adverse event (AE), including 2 with grade 5 (death) and 2 with grade 4 (disabling) infectious AE (median follow-up of 1.65 years [range 0.1-8.5]). No patients developed progressive multifocal leukoencephalopathy. A definite, probable, or possible benefit was reported in 125 of 144 (87%) patients. A total of 17.4% of patients had a modified Rankin Scale (mRS) score of 0-2 at rituximab initiation, compared to 73.9% at outcome. The change in mRS 0-2 was greater in patients given rituximab early in their disease course compared to those treated later. Conclusion: While limited by the retrospective nature of this analysis, our data support an off-label use of rituximab, although the significant risk of infectious complications suggests rituximab should be restricted to disorders with significant morbidity and mortality. Classification of evidence: This study provides Class IV evidence that in pediatric autoimmune and inflammatory CNS disorders, rituximab improves neurologic outcomes with a 7.6% risk of adverse infections.

Affiliations:

Australie, Canada, France, Royaume-Uni, Turquie, États-Unis
Alberta, Angleterre, Grand Londres, Ontario, Oxfordshire, Pennsylvanie, Texas
Calgary, Houston, Londres, Oxford, Pittsburgh, Toronto
Université de Calgary, Université de Pittsburgh, Université de Toronto

Links toward previous steps (curation, corpus...)

to stream PascalFrancis, to step Corpus: 000846
to stream PascalFrancis, to step Curation: 003D85
to stream PascalFrancis, to step Checkpoint: 000050
to stream Main, to step Merge: 004F73
to stream Main, to step Curation: 004D32
to stream Main, to step Exploration: 004D32
to stream France, to step Extraction: 000311

Links to Exploration step

Pascal:14-0183455

Le document en format XML

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<author><name sortKey="Alper, Gulay" sort="Alper, Gulay" uniqKey="Alper G" first="Gulay" last="Alper">Gulay Alper</name>
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<author><name sortKey="Riney, Catherine J" sort="Riney, Catherine J" uniqKey="Riney C" first="Catherine J." last="Riney">Catherine J. Riney</name>
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<author><name sortKey="Kornberg, Andrew" sort="Kornberg, Andrew" uniqKey="Kornberg A" first="Andrew" last="Kornberg">Andrew Kornberg</name>
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<author><name sortKey="Tardieu, Marc" sort="Tardieu, Marc" uniqKey="Tardieu M" first="Marc" last="Tardieu">Marc Tardieu</name>
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<author><name sortKey="Stocco, Amber" sort="Stocco, Amber" uniqKey="Stocco A" first="Amber" last="Stocco">Amber Stocco</name>
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<author><name sortKey="Banwell, Brenda" sort="Banwell, Brenda" uniqKey="Banwell B" first="Brenda" last="Banwell">Brenda Banwell</name>
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<wicri:noRegion>Children's Hospital of Philadelphia, University of Pennsylvania</wicri:noRegion>
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<author><name sortKey="Gorman, Mark P" sort="Gorman, Mark P" uniqKey="Gorman M" first="Mark P." last="Gorman">Mark P. Gorman</name>
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<s3>CAN</s3>
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<country>Canada</country>
<wicri:noRegion>Boston Children's Hospital</wicri:noRegion>
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<author><name sortKey="Benseler, Susanne M" sort="Benseler, Susanne M" uniqKey="Benseler S" first="Susanne M." last="Benseler">Susanne M. Benseler</name>
<affiliation wicri:level="4"><inist:fA14 i1="03"><s1>Division of Rheumatology, The Hospital for Sick Children, University of Toronto</s1>
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<region type="state">Ontario</region>
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<orgName type="university">Université de Toronto</orgName>
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<affiliation wicri:level="4"><inist:fA14 i1="16"><s1>Alberta Children's Hospital, Calgary, University of Calgary</s1>
<s3>CAN</s3>
<sZ>25 aut.</sZ>
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<country>Canada</country>
<placeName><settlement type="city">Calgary</settlement>
<region type="state">Alberta</region>
</placeName>
<orgName type="university">Université de Calgary</orgName>
</affiliation>
</author>
<author><name sortKey="Ming Lim" sort="Ming Lim" uniqKey="Ming Lim" last="Ming Lim">MING LIM</name>
<affiliation wicri:level="3"><inist:fA14 i1="17"><s1>Evelina Children's Hospital</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>26 aut.</sZ>
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<country>Royaume-Uni</country>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
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<sourceDesc><biblStruct><analytic><title xml:lang="en" level="a">Utility and safety of rituximab in pediatric autoimmune and inflammatory CNS disease</title>
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<affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Neuroimmunology Group, Children's Hospital at Westmead, University of Sydney</s1>
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<author><name sortKey="Brilot, Fabienne" sort="Brilot, Fabienne" uniqKey="Brilot F" first="Fabienne" last="Brilot">Fabienne Brilot</name>
<affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Neuroimmunology Group, Children's Hospital at Westmead, University of Sydney</s1>
<s3>AUS</s3>
<sZ>1 aut.</sZ>
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<author><name sortKey="Duffy, Lisa V" sort="Duffy, Lisa V" uniqKey="Duffy L" first="Lisa V." last="Duffy">Lisa V. Duffy</name>
<affiliation wicri:level="1"><inist:fA14 i1="02"><s1>Boston Children's Hospital</s1>
<s2>MA</s2>
<s3>CAN</s3>
<sZ>3 aut.</sZ>
<sZ>24 aut.</sZ>
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<country>Canada</country>
<wicri:noRegion>Boston Children's Hospital</wicri:noRegion>
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<author><name sortKey="Twilt, Marinka" sort="Twilt, Marinka" uniqKey="Twilt M" first="Marinka" last="Twilt">Marinka Twilt</name>
<affiliation wicri:level="4"><inist:fA14 i1="03"><s1>Division of Rheumatology, The Hospital for Sick Children, University of Toronto</s1>
<s3>CAN</s3>
<sZ>4 aut.</sZ>
<sZ>25 aut.</sZ>
</inist:fA14>
<country>Canada</country>
<placeName><settlement type="city">Toronto</settlement>
<region type="state">Ontario</region>
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<orgName type="university">Université de Toronto</orgName>
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<author><name sortKey="Waldman, Amy T" sort="Waldman, Amy T" uniqKey="Waldman A" first="Amy T." last="Waldman">Amy T. Waldman</name>
<affiliation wicri:level="1"><inist:fA14 i1="04"><s1>Children's Hospital of Philadelphia, University of Pennsylvania</s1>
<s3>USA</s3>
<sZ>5 aut.</sZ>
<sZ>6 aut.</sZ>
<sZ>23 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<wicri:noRegion>Children's Hospital of Philadelphia, University of Pennsylvania</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Narula, Sona" sort="Narula, Sona" uniqKey="Narula S" first="Sona" last="Narula">Sona Narula</name>
<affiliation wicri:level="1"><inist:fA14 i1="04"><s1>Children's Hospital of Philadelphia, University of Pennsylvania</s1>
<s3>USA</s3>
<sZ>5 aut.</sZ>
<sZ>6 aut.</sZ>
<sZ>23 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<wicri:noRegion>Children's Hospital of Philadelphia, University of Pennsylvania</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Muscal, Eyal" sort="Muscal, Eyal" uniqKey="Muscal E" first="Eyal" last="Muscal">Eyal Muscal</name>
<affiliation wicri:level="3"><inist:fA14 i1="05"><s1>Paediatric Neurology and Rheumatology, Texas Children's Hospital, Baylor College of Medicine</s1>
<s2>Houston</s2>
<s3>USA</s3>
<sZ>7 aut.</sZ>
<sZ>22 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<placeName><settlement type="city">Houston</settlement>
<region type="state">Texas</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Deiva, Kumaran" sort="Deiva, Kumaran" uniqKey="Deiva K" first="Kumaran" last="Deiva">Kumaran Deiva</name>
<affiliation wicri:level="1"><inist:fA14 i1="06"><s1>Assistance Publique-Hopitaux de Paris, Hôpitaux Universitaires Paris-Sud, National Referral Center for Neuro-Inflammatory Diseases in Children, Pediatric Neurology Department and Université Paris-Sud, Inserm U1012</s1>
<s2>Le Kremlin-Bicêtre</s2>
<s3>FRA</s3>
<sZ>8 aut.</sZ>
<sZ>21 aut.</sZ>
</inist:fA14>
<country>France</country>
<wicri:noRegion>Le Kremlin-Bicêtre</wicri:noRegion>
<wicri:noRegion>Inserm U1012</wicri:noRegion>
<wicri:noRegion>Le Kremlin-Bicêtre</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Andersen, Erik" sort="Andersen, Erik" uniqKey="Andersen E" first="Erik" last="Andersen">Erik Andersen</name>
<affiliation wicri:level="1"><inist:fA14 i1="07"><s1>Royal Children's Hospital Melbourne</s1>
<s3>AUS</s3>
<sZ>9 aut.</sZ>
<sZ>20 aut.</sZ>
</inist:fA14>
<country>Australie</country>
<wicri:noRegion>Royal Children's Hospital Melbourne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Eyre, Michael R" sort="Eyre, Michael R" uniqKey="Eyre M" first="Michael R." last="Eyre">Michael R. Eyre</name>
<affiliation wicri:level="3"><inist:fA14 i1="08"><s1>Great Ormond Street Hospital NHS Trust</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>10 aut.</sZ>
<sZ>18 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Eleftheriou, Despina" sort="Eleftheriou, Despina" uniqKey="Eleftheriou D" first="Despina" last="Eleftheriou">Despina Eleftheriou</name>
<affiliation wicri:level="3"><inist:fA14 i1="09"><s1>UCL Institute of Child Health</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>11 aut.</sZ>
<sZ>12 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Brogan, Paul A" sort="Brogan, Paul A" uniqKey="Brogan P" first="Paul A." last="Brogan">Paul A. Brogan</name>
<affiliation wicri:level="3"><inist:fA14 i1="09"><s1>UCL Institute of Child Health</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>11 aut.</sZ>
<sZ>12 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Kneen, Rachel" sort="Kneen, Rachel" uniqKey="Kneen R" first="Rachel" last="Kneen">Rachel Kneen</name>
<affiliation wicri:level="1"><inist:fA14 i1="10"><s1>Alder Hey Children's NHS Foundation Trust and the Institute of Infection and Global Health, University of Liverpool</s1>
<s3>GBR</s3>
<sZ>13 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<wicri:noRegion>Alder Hey Children's NHS Foundation Trust and the Institute of Infection and Global Health, University of Liverpool</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Alper, Gulay" sort="Alper, Gulay" uniqKey="Alper G" first="Gulay" last="Alper">Gulay Alper</name>
<affiliation wicri:level="4"><inist:fA14 i1="11"><s1>Children's Hospital of Pittsburgh, University of Pittsburgh</s1>
<s2>PA</s2>
<s3>USA</s3>
<sZ>14 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<placeName><settlement type="city">Pittsburgh</settlement>
<region type="state">Pennsylvanie</region>
</placeName>
<orgName type="university">Université de Pittsburgh</orgName>
</affiliation>
</author>
<author><name sortKey="Anlar, Banu" sort="Anlar, Banu" uniqKey="Anlar B" first="Banu" last="Anlar">Banu Anlar</name>
<affiliation wicri:level="1"><inist:fA14 i1="12"><s1>Hacettepe University Child Neurology</s1>
<s3>TUR</s3>
<sZ>15 aut.</sZ>
</inist:fA14>
<country>Turquie</country>
<wicri:noRegion>Hacettepe University Child Neurology</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Wassmer, Evangeline" sort="Wassmer, Evangeline" uniqKey="Wassmer E" first="Evangeline" last="Wassmer">Evangeline Wassmer</name>
<affiliation wicri:level="1"><inist:fA14 i1="13"><s1>Birmingham Children's Hospital</s1>
<s3>USA</s3>
<sZ>16 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<wicri:noRegion>Birmingham Children's Hospital</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Heineman, Kirsten" sort="Heineman, Kirsten" uniqKey="Heineman K" first="Kirsten" last="Heineman">Kirsten Heineman</name>
<affiliation wicri:level="3"><inist:fA14 i1="14"><s1>John Radcliffe Hospital</s1>
<s2>Oxford</s2>
<s3>GBR</s3>
<sZ>17 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<placeName><settlement type="city">Oxford</settlement>
<region type="country">Angleterre</region>
<region type="comté" nuts="2">Oxfordshire</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Hemingway, Cheryl" sort="Hemingway, Cheryl" uniqKey="Hemingway C" first="Cheryl" last="Hemingway">Cheryl Hemingway</name>
<affiliation wicri:level="3"><inist:fA14 i1="08"><s1>Great Ormond Street Hospital NHS Trust</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>10 aut.</sZ>
<sZ>18 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Riney, Catherine J" sort="Riney, Catherine J" uniqKey="Riney C" first="Catherine J." last="Riney">Catherine J. Riney</name>
<affiliation wicri:level="1"><inist:fA14 i1="15"><s1>Neurosciences Unit and Mater Medical Research Institute), Mater Children's Hospital, South Brisbane, University of Queensland</s1>
<s3>AUS</s3>
<sZ>19 aut.</sZ>
</inist:fA14>
<country>Australie</country>
<wicri:noRegion>Neurosciences Unit and Mater Medical Research Institute), Mater Children's Hospital, South Brisbane, University of Queensland</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Kornberg, Andrew" sort="Kornberg, Andrew" uniqKey="Kornberg A" first="Andrew" last="Kornberg">Andrew Kornberg</name>
<affiliation wicri:level="1"><inist:fA14 i1="07"><s1>Royal Children's Hospital Melbourne</s1>
<s3>AUS</s3>
<sZ>9 aut.</sZ>
<sZ>20 aut.</sZ>
</inist:fA14>
<country>Australie</country>
<wicri:noRegion>Royal Children's Hospital Melbourne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Tardieu, Marc" sort="Tardieu, Marc" uniqKey="Tardieu M" first="Marc" last="Tardieu">Marc Tardieu</name>
<affiliation wicri:level="1"><inist:fA14 i1="06"><s1>Assistance Publique-Hopitaux de Paris, Hôpitaux Universitaires Paris-Sud, National Referral Center for Neuro-Inflammatory Diseases in Children, Pediatric Neurology Department and Université Paris-Sud, Inserm U1012</s1>
<s2>Le Kremlin-Bicêtre</s2>
<s3>FRA</s3>
<sZ>8 aut.</sZ>
<sZ>21 aut.</sZ>
</inist:fA14>
<country>France</country>
<wicri:noRegion>Le Kremlin-Bicêtre</wicri:noRegion>
<wicri:noRegion>Inserm U1012</wicri:noRegion>
<wicri:noRegion>Le Kremlin-Bicêtre</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Stocco, Amber" sort="Stocco, Amber" uniqKey="Stocco A" first="Amber" last="Stocco">Amber Stocco</name>
<affiliation wicri:level="3"><inist:fA14 i1="05"><s1>Paediatric Neurology and Rheumatology, Texas Children's Hospital, Baylor College of Medicine</s1>
<s2>Houston</s2>
<s3>USA</s3>
<sZ>7 aut.</sZ>
<sZ>22 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<placeName><settlement type="city">Houston</settlement>
<region type="state">Texas</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Banwell, Brenda" sort="Banwell, Brenda" uniqKey="Banwell B" first="Brenda" last="Banwell">Brenda Banwell</name>
<affiliation wicri:level="1"><inist:fA14 i1="04"><s1>Children's Hospital of Philadelphia, University of Pennsylvania</s1>
<s3>USA</s3>
<sZ>5 aut.</sZ>
<sZ>6 aut.</sZ>
<sZ>23 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
<wicri:noRegion>Children's Hospital of Philadelphia, University of Pennsylvania</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Gorman, Mark P" sort="Gorman, Mark P" uniqKey="Gorman M" first="Mark P." last="Gorman">Mark P. Gorman</name>
<affiliation wicri:level="1"><inist:fA14 i1="02"><s1>Boston Children's Hospital</s1>
<s2>MA</s2>
<s3>CAN</s3>
<sZ>3 aut.</sZ>
<sZ>24 aut.</sZ>
</inist:fA14>
<country>Canada</country>
<wicri:noRegion>Boston Children's Hospital</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Benseler, Susanne M" sort="Benseler, Susanne M" uniqKey="Benseler S" first="Susanne M." last="Benseler">Susanne M. Benseler</name>
<affiliation wicri:level="4"><inist:fA14 i1="03"><s1>Division of Rheumatology, The Hospital for Sick Children, University of Toronto</s1>
<s3>CAN</s3>
<sZ>4 aut.</sZ>
<sZ>25 aut.</sZ>
</inist:fA14>
<country>Canada</country>
<placeName><settlement type="city">Toronto</settlement>
<region type="state">Ontario</region>
</placeName>
<orgName type="university">Université de Toronto</orgName>
</affiliation>
<affiliation wicri:level="4"><inist:fA14 i1="16"><s1>Alberta Children's Hospital, Calgary, University of Calgary</s1>
<s3>CAN</s3>
<sZ>25 aut.</sZ>
</inist:fA14>
<country>Canada</country>
<placeName><settlement type="city">Calgary</settlement>
<region type="state">Alberta</region>
</placeName>
<orgName type="university">Université de Calgary</orgName>
</affiliation>
</author>
<author><name sortKey="Ming Lim" sort="Ming Lim" uniqKey="Ming Lim" last="Ming Lim">MING LIM</name>
<affiliation wicri:level="3"><inist:fA14 i1="17"><s1>Evelina Children's Hospital</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>26 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
</placeName>
</affiliation>
</author>
</analytic>
<series><title level="j" type="main">Neurology</title>
<title level="j" type="abbreviated">Neurology</title>
<idno type="ISSN">0028-3878</idno>
<imprint><date when="2014">2014</date>
</imprint>
</series>
</biblStruct>
</sourceDesc>
<seriesStmt><title level="j" type="main">Neurology</title>
<title level="j" type="abbreviated">Neurology</title>
<idno type="ISSN">0028-3878</idno>
</seriesStmt>
</fileDesc>
<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Autoimmune disease</term>
<term>Central nervous system</term>
<term>Child</term>
<term>Inflammatory disease</term>
<term>Nervous system diseases</term>
<term>Pediatrics</term>
<term>Rituximab</term>
<term>Safety</term>
</keywords>
<keywords scheme="Pascal" xml:lang="fr"><term>Pathologie du   système nerveux</term>
<term>Sécurité</term>
<term>Rituximab</term>
<term>Enfant</term>
<term>Pédiatrie</term>
<term>Maladie autoimmune</term>
<term>Maladie inflammatoire</term>
<term>Système nerveux central</term>
</keywords>
<keywords scheme="Wicri" type="topic" xml:lang="fr"><term>Enfant</term>
<term>Pédiatrie</term>
</keywords>
</textClass>
</profileDesc>
</teiHeader>
<front><div type="abstract" xml:lang="en">Objective: To assess the utility and safety of rituximab in pediatric autoimmune and inflammatory disorders of the CNS. Methods: Multicenter retrospective study. Results: A total of 144 children and adolescents (median age 8 years, range 0.7-17; 103 female) with NMDA receptor (NMDAR) encephalitis (n = 39), opsoclonus myoclonus ataxia syndrome (n = 32), neuromyelitis optica spectrum disorders (n = 20), neuropsychiatric systemic lupus erythematosus (n = 18), and other neuroinflammatory disorders (n = 35) were studied. Rituximab was given after a median duration of disease of 0.5 years (range 0.05-9.5 years). Infusion adverse events were recorded in 18/144 (12.5%), including grade 4 (anaphylaxis) in 3. Eleven patients (7.6%) had an infectious adverse event (AE), including 2 with grade 5 (death) and 2 with grade 4 (disabling) infectious AE (median follow-up of 1.65 years [range 0.1-8.5]). No patients developed progressive multifocal leukoencephalopathy. A definite, probable, or possible benefit was reported in 125 of 144 (87%) patients. A total of 17.4% of patients had a modified Rankin Scale (mRS) score of 0-2 at rituximab initiation, compared to 73.9% at outcome. The change in mRS 0-2 was greater in patients given rituximab early in their disease course compared to those treated later. Conclusion: While limited by the retrospective nature of this analysis, our data support an off-label use of rituximab, although the significant risk of infectious complications suggests rituximab should be restricted to disorders with significant morbidity and mortality. Classification of evidence: This study provides Class IV evidence that in pediatric autoimmune and inflammatory CNS disorders, rituximab improves neurologic outcomes with a 7.6% risk of adverse infections.</div>
</front>
</TEI>
<affiliations><list><country><li>Australie</li>
<li>Canada</li>
<li>France</li>
<li>Royaume-Uni</li>
<li>Turquie</li>
<li>États-Unis</li>
</country>
<region><li>Alberta</li>
<li>Angleterre</li>
<li>Grand Londres</li>
<li>Ontario</li>
<li>Oxfordshire</li>
<li>Pennsylvanie</li>
<li>Texas</li>
</region>
<settlement><li>Calgary</li>
<li>Houston</li>
<li>Londres</li>
<li>Oxford</li>
<li>Pittsburgh</li>
<li>Toronto</li>
</settlement>
<orgName><li>Université de Calgary</li>
<li>Université de Pittsburgh</li>
<li>Université de Toronto</li>
</orgName>
</list>
<tree><country name="Australie"><noRegion><name sortKey="Dale, Russell C" sort="Dale, Russell C" uniqKey="Dale R" first="Russell C." last="Dale">Russell C. Dale</name>
</noRegion>
<name sortKey="Andersen, Erik" sort="Andersen, Erik" uniqKey="Andersen E" first="Erik" last="Andersen">Erik Andersen</name>
<name sortKey="Brilot, Fabienne" sort="Brilot, Fabienne" uniqKey="Brilot F" first="Fabienne" last="Brilot">Fabienne Brilot</name>
<name sortKey="Kornberg, Andrew" sort="Kornberg, Andrew" uniqKey="Kornberg A" first="Andrew" last="Kornberg">Andrew Kornberg</name>
<name sortKey="Riney, Catherine J" sort="Riney, Catherine J" uniqKey="Riney C" first="Catherine J." last="Riney">Catherine J. Riney</name>
</country>
<country name="Canada"><noRegion><name sortKey="Duffy, Lisa V" sort="Duffy, Lisa V" uniqKey="Duffy L" first="Lisa V." last="Duffy">Lisa V. Duffy</name>
</noRegion>
<name sortKey="Benseler, Susanne M" sort="Benseler, Susanne M" uniqKey="Benseler S" first="Susanne M." last="Benseler">Susanne M. Benseler</name>
<name sortKey="Benseler, Susanne M" sort="Benseler, Susanne M" uniqKey="Benseler S" first="Susanne M." last="Benseler">Susanne M. Benseler</name>
<name sortKey="Gorman, Mark P" sort="Gorman, Mark P" uniqKey="Gorman M" first="Mark P." last="Gorman">Mark P. Gorman</name>
<name sortKey="Twilt, Marinka" sort="Twilt, Marinka" uniqKey="Twilt M" first="Marinka" last="Twilt">Marinka Twilt</name>
</country>
<country name="États-Unis"><noRegion><name sortKey="Waldman, Amy T" sort="Waldman, Amy T" uniqKey="Waldman A" first="Amy T." last="Waldman">Amy T. Waldman</name>
</noRegion>
<name sortKey="Alper, Gulay" sort="Alper, Gulay" uniqKey="Alper G" first="Gulay" last="Alper">Gulay Alper</name>
<name sortKey="Banwell, Brenda" sort="Banwell, Brenda" uniqKey="Banwell B" first="Brenda" last="Banwell">Brenda Banwell</name>
<name sortKey="Muscal, Eyal" sort="Muscal, Eyal" uniqKey="Muscal E" first="Eyal" last="Muscal">Eyal Muscal</name>
<name sortKey="Narula, Sona" sort="Narula, Sona" uniqKey="Narula S" first="Sona" last="Narula">Sona Narula</name>
<name sortKey="Stocco, Amber" sort="Stocco, Amber" uniqKey="Stocco A" first="Amber" last="Stocco">Amber Stocco</name>
<name sortKey="Wassmer, Evangeline" sort="Wassmer, Evangeline" uniqKey="Wassmer E" first="Evangeline" last="Wassmer">Evangeline Wassmer</name>
</country>
<country name="France"><noRegion><name sortKey="Deiva, Kumaran" sort="Deiva, Kumaran" uniqKey="Deiva K" first="Kumaran" last="Deiva">Kumaran Deiva</name>
</noRegion>
<name sortKey="Tardieu, Marc" sort="Tardieu, Marc" uniqKey="Tardieu M" first="Marc" last="Tardieu">Marc Tardieu</name>
</country>
<country name="Royaume-Uni"><region name="Angleterre"><name sortKey="Eyre, Michael R" sort="Eyre, Michael R" uniqKey="Eyre M" first="Michael R." last="Eyre">Michael R. Eyre</name>
</region>
<name sortKey="Brogan, Paul A" sort="Brogan, Paul A" uniqKey="Brogan P" first="Paul A." last="Brogan">Paul A. Brogan</name>
<name sortKey="Eleftheriou, Despina" sort="Eleftheriou, Despina" uniqKey="Eleftheriou D" first="Despina" last="Eleftheriou">Despina Eleftheriou</name>
<name sortKey="Heineman, Kirsten" sort="Heineman, Kirsten" uniqKey="Heineman K" first="Kirsten" last="Heineman">Kirsten Heineman</name>
<name sortKey="Hemingway, Cheryl" sort="Hemingway, Cheryl" uniqKey="Hemingway C" first="Cheryl" last="Hemingway">Cheryl Hemingway</name>
<name sortKey="Kneen, Rachel" sort="Kneen, Rachel" uniqKey="Kneen R" first="Rachel" last="Kneen">Rachel Kneen</name>
<name sortKey="Ming Lim" sort="Ming Lim" uniqKey="Ming Lim" last="Ming Lim">MING LIM</name>
</country>
<country name="Turquie"><noRegion><name sortKey="Anlar, Banu" sort="Anlar, Banu" uniqKey="Anlar B" first="Banu" last="Anlar">Banu Anlar</name>
</noRegion>
</country>
</tree>
</affiliations>
</record>

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Utility and safety of rituximab in pediatric autoimmune and inflammatory CNS disease

Utility and safety of rituximab in pediatric autoimmune and inflammatory CNS disease

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