PittsburghV1, PascalFrancis, Checkpoint, bibRecord, 000050

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

Identifieur interne : 000050 ( PascalFrancis/Checkpoint ); précédent : 000049; suivant : 000051

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

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Pascal:14-0183455

Le document en format XML

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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>
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<author><name sortKey="Muscal, Eyal" sort="Muscal, Eyal" uniqKey="Muscal E" first="Eyal" last="Muscal">Eyal Muscal</name>
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<author><name sortKey="Deiva, Kumaran" sort="Deiva, Kumaran" uniqKey="Deiva K" first="Kumaran" last="Deiva">Kumaran Deiva</name>
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<author><name sortKey="Andersen, Erik" sort="Andersen, Erik" uniqKey="Andersen E" first="Erik" last="Andersen">Erik Andersen</name>
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<author><name sortKey="Eyre, Michael R" sort="Eyre, Michael R" uniqKey="Eyre M" first="Michael R." last="Eyre">Michael R. Eyre</name>
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<author><name sortKey="Brogan, Paul A" sort="Brogan, Paul A" uniqKey="Brogan P" first="Paul A." last="Brogan">Paul A. Brogan</name>
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<author><name sortKey="Kneen, Rachel" sort="Kneen, Rachel" uniqKey="Kneen R" first="Rachel" last="Kneen">Rachel Kneen</name>
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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="Anlar, Banu" sort="Anlar, Banu" uniqKey="Anlar B" first="Banu" last="Anlar">Banu Anlar</name>
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<country>Turquie</country>
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<author><name sortKey="Wassmer, Evangeline" sort="Wassmer, Evangeline" uniqKey="Wassmer E" first="Evangeline" last="Wassmer">Evangeline Wassmer</name>
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<author><name sortKey="Heineman, Kirsten" sort="Heineman, Kirsten" uniqKey="Heineman K" first="Kirsten" last="Heineman">Kirsten Heineman</name>
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<author><name sortKey="Hemingway, Cheryl" sort="Hemingway, Cheryl" uniqKey="Hemingway C" first="Cheryl" last="Hemingway">Cheryl Hemingway</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>
<affiliation wicri:level="1"><inist:fA14 i1="04"><s1>Children's Hospital of Philadelphia, University of Pennsylvania</s1>
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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>
<affiliation wicri:level="1"><inist:fA14 i1="02"><s1>Boston Children's Hospital</s1>
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<s3>CAN</s3>
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<country>Canada</country>
<wicri:noRegion>Boston Children's Hospital</wicri:noRegion>
</affiliation>
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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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<placeName><settlement type="city">Toronto</settlement>
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</placeName>
<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>
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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>
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<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>
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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>
<author><name sortKey="Dale, Russell C" sort="Dale, Russell C" uniqKey="Dale R" first="Russell C." last="Dale">Russell C. Dale</name>
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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>
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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>
</inist:fA14>
<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>
</placeName>
<orgName type="university">Université de Toronto</orgName>
</affiliation>
</author>
<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>
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<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>
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</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>
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<fA08 i1="01" i2="1" l="ENG"><s1>Utility and safety of rituximab in pediatric autoimmune and inflammatory CNS disease</s1>
</fA08>
<fA11 i1="01" i2="1"><s1>DALE (Russell C.)</s1>
</fA11>
<fA11 i1="02" i2="1"><s1>BRILOT (Fabienne)</s1>
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<fA11 i1="10" i2="1"><s1>EYRE (Michael R.)</s1>
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<fA11 i1="11" i2="1"><s1>ELEFTHERIOU (Despina)</s1>
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<fA11 i1="12" i2="1"><s1>BROGAN (Paul A.)</s1>
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<fA11 i1="13" i2="1"><s1>KNEEN (Rachel)</s1>
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<fA11 i1="14" i2="1"><s1>ALPER (Gulay)</s1>
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<fA11 i1="15" i2="1"><s1>ANLAR (Banu)</s1>
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<fA11 i1="16" i2="1"><s1>WASSMER (Evangeline)</s1>
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<fA11 i1="17" i2="1"><s1>HEINEMAN (Kirsten)</s1>
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<fA11 i1="18" i2="1"><s1>HEMINGWAY (Cheryl)</s1>
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<fA11 i1="19" i2="1"><s1>RINEY (Catherine J.)</s1>
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<fA11 i1="20" i2="1"><s1>KORNBERG (Andrew)</s1>
</fA11>
<fA11 i1="21" i2="1"><s1>TARDIEU (Marc)</s1>
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<fA11 i1="22" i2="1"><s1>STOCCO (Amber)</s1>
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<fA11 i1="23" i2="1"><s1>BANWELL (Brenda)</s1>
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<fA11 i1="24" i2="1"><s1>GORMAN (Mark P.)</s1>
</fA11>
<fA11 i1="25" i2="1"><s1>BENSELER (Susanne M.)</s1>
</fA11>
<fA11 i1="26" i2="1"><s1>MING LIM</s1>
</fA11>
<fA14 i1="01"><s1>Neuroimmunology Group, Children's Hospital at Westmead, University of Sydney</s1>
<s3>AUS</s3>
<sZ>1 aut.</sZ>
<sZ>2 aut.</sZ>
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<fA14 i1="02"><s1>Boston Children's Hospital</s1>
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<s3>CAN</s3>
<sZ>3 aut.</sZ>
<sZ>24 aut.</sZ>
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<fA14 i1="03"><s1>Division of Rheumatology, The Hospital for Sick Children, University of Toronto</s1>
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<sZ>4 aut.</sZ>
<sZ>25 aut.</sZ>
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<fA14 i1="04"><s1>Children's Hospital of Philadelphia, University of Pennsylvania</s1>
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<sZ>5 aut.</sZ>
<sZ>6 aut.</sZ>
<sZ>23 aut.</sZ>
</fA14>
<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>
</fA14>
<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>
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<fA14 i1="07"><s1>Royal Children's Hospital Melbourne</s1>
<s3>AUS</s3>
<sZ>9 aut.</sZ>
<sZ>20 aut.</sZ>
</fA14>
<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>
</fA14>
<fA14 i1="09"><s1>UCL Institute of Child Health</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>11 aut.</sZ>
<sZ>12 aut.</sZ>
</fA14>
<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>
</fA14>
<fA14 i1="11"><s1>Children's Hospital of Pittsburgh, University of Pittsburgh</s1>
<s2>PA</s2>
<s3>USA</s3>
<sZ>14 aut.</sZ>
</fA14>
<fA14 i1="12"><s1>Hacettepe University Child Neurology</s1>
<s3>TUR</s3>
<sZ>15 aut.</sZ>
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<fA14 i1="13"><s1>Birmingham Children's Hospital</s1>
<s3>USA</s3>
<sZ>16 aut.</sZ>
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<fA14 i1="14"><s1>John Radcliffe Hospital</s1>
<s2>Oxford</s2>
<s3>GBR</s3>
<sZ>17 aut.</sZ>
</fA14>
<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>
</fA14>
<fA14 i1="16"><s1>Alberta Children's Hospital, Calgary, University of Calgary</s1>
<s3>CAN</s3>
<sZ>25 aut.</sZ>
</fA14>
<fA14 i1="17"><s1>Evelina Children's Hospital</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>26 aut.</sZ>
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<fC01 i1="01" l="ENG"><s0>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.</s0>
</fC01>
<fC02 i1="01" i2="X"><s0>002B17</s0>
</fC02>
<fC02 i1="02" i2="X"><s0>002B17G</s0>
</fC02>
<fC03 i1="01" i2="X" l="FRE"><s0>Pathologie du   système nerveux</s0>
<s5>01</s5>
</fC03>
<fC03 i1="01" i2="X" l="ENG"><s0>Nervous system diseases</s0>
<s5>01</s5>
</fC03>
<fC03 i1="01" i2="X" l="SPA"><s0>Sistema nervioso patología</s0>
<s5>01</s5>
</fC03>
<fC03 i1="02" i2="X" l="FRE"><s0>Sécurité</s0>
<s5>09</s5>
</fC03>
<fC03 i1="02" i2="X" l="ENG"><s0>Safety</s0>
<s5>09</s5>
</fC03>
<fC03 i1="02" i2="X" l="SPA"><s0>Seguridad</s0>
<s5>09</s5>
</fC03>
<fC03 i1="03" i2="X" l="FRE"><s0>Rituximab</s0>
<s2>NK</s2>
<s2>FR</s2>
<s5>10</s5>
</fC03>
<fC03 i1="03" i2="X" l="ENG"><s0>Rituximab</s0>
<s2>NK</s2>
<s2>FR</s2>
<s5>10</s5>
</fC03>
<fC03 i1="03" i2="X" l="SPA"><s0>Rituximab</s0>
<s2>NK</s2>
<s2>FR</s2>
<s5>10</s5>
</fC03>
<fC03 i1="04" i2="X" l="FRE"><s0>Enfant</s0>
<s5>11</s5>
</fC03>
<fC03 i1="04" i2="X" l="ENG"><s0>Child</s0>
<s5>11</s5>
</fC03>
<fC03 i1="04" i2="X" l="SPA"><s0>Niño</s0>
<s5>11</s5>
</fC03>
<fC03 i1="05" i2="X" l="FRE"><s0>Pédiatrie</s0>
<s5>12</s5>
</fC03>
<fC03 i1="05" i2="X" l="ENG"><s0>Pediatrics</s0>
<s5>12</s5>
</fC03>
<fC03 i1="05" i2="X" l="SPA"><s0>Pediatría</s0>
<s5>12</s5>
</fC03>
<fC03 i1="06" i2="X" l="FRE"><s0>Maladie autoimmune</s0>
<s5>13</s5>
</fC03>
<fC03 i1="06" i2="X" l="ENG"><s0>Autoimmune disease</s0>
<s5>13</s5>
</fC03>
<fC03 i1="06" i2="X" l="SPA"><s0>Enfermedad autoinmune</s0>
<s5>13</s5>
</fC03>
<fC03 i1="07" i2="X" l="FRE"><s0>Maladie inflammatoire</s0>
<s5>14</s5>
</fC03>
<fC03 i1="07" i2="X" l="ENG"><s0>Inflammatory disease</s0>
<s5>14</s5>
</fC03>
<fC03 i1="07" i2="X" l="SPA"><s0>Enfermedad inflamatoria</s0>
<s5>14</s5>
</fC03>
<fC03 i1="08" i2="X" l="FRE"><s0>Système nerveux central</s0>
<s5>15</s5>
</fC03>
<fC03 i1="08" i2="X" l="ENG"><s0>Central nervous system</s0>
<s5>15</s5>
</fC03>
<fC03 i1="08" i2="X" l="SPA"><s0>Sistema nervioso central</s0>
<s5>15</s5>
</fC03>
<fC07 i1="01" i2="X" l="FRE"><s0>Homme</s0>
</fC07>
<fC07 i1="01" i2="X" l="ENG"><s0>Human</s0>
</fC07>
<fC07 i1="01" i2="X" l="SPA"><s0>Hombre</s0>
</fC07>
<fC07 i1="02" i2="X" l="FRE"><s0>Immunopathologie</s0>
<s5>37</s5>
</fC07>
<fC07 i1="02" i2="X" l="ENG"><s0>Immunopathology</s0>
<s5>37</s5>
</fC07>
<fC07 i1="02" i2="X" l="SPA"><s0>Inmunopatología</s0>
<s5>37</s5>
</fC07>
<fN21><s1>230</s1>
</fN21>
<fN44 i1="01"><s1>OTO</s1>
</fN44>
<fN82><s1>OTO</s1>
</fN82>
</pA>
</standard>
</inist>
<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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