B cell depletion therapy for new‐onset opsoclonus‐myoclonus
Identifieur interne : 002276 ( Main/Merge ); précédent : 002275; suivant : 002277B cell depletion therapy for new‐onset opsoclonus‐myoclonus
Auteurs : Michael R. Pranzatelli ; Elizabeth D. Tate ; Jennifer A. Swan ; Anna L. Travelstead ; Jerry A. Colliver [États-Unis] ; Steven J. Verhulst [États-Unis] ; Carl J. Crosley [États-Unis] ; William D. Graf [États-Unis] ; Suja A. Joseph [États-Unis] ; Howard M. Kelfer [États-Unis] ; G. Praveen Raju [États-Unis]Source :
- Movement Disorders [ 0885-3185 ] ; 2010-01-30.
English descriptors
- KwdEn :
- ACTH, Adrenocorticotropic Hormone (adverse effects), Adrenocorticotropic Hormone (therapeutic use), Analysis of Variance, Antibodies, Monoclonal (adverse effects), Antibodies, Monoclonal (therapeutic use), Antibodies, Monoclonal, Murine-Derived, Antineoplastic Combined Chemotherapy Protocols (adverse effects), Antineoplastic Combined Chemotherapy Protocols (therapeutic use), Ataxia (drug therapy), B-Lymphocytes (drug effects), B-Lymphocytes (immunology), B-Lymphocytes (pathology), Child, Preschool, Drug Administration Schedule, Drug Therapy, Combination (methods), Female, Humans, Immunoglobulin M (blood), Immunoglobulins (adverse effects), Immunoglobulins (therapeutic use), Immunologic Factors (adverse effects), Immunologic Factors (therapeutic use), Infant, Kinsbourne syndrome, Lymphocyte Depletion (methods), Male, Myoclonus (drug therapy), Opsoclonus-Myoclonus Syndrome (physiopathology), Opsoclonus-Myoclonus Syndrome (therapy), Treatment Outcome, anti‐B cell agent, dancing eyes, neuroblastoma, paraneoplastic syndrome, rituximab.
- MESH :
- chemical , adverse effects : Adrenocorticotropic Hormone, Antibodies, Monoclonal, Immunoglobulins, Immunologic Factors.
- chemical , blood : Immunoglobulin M.
- chemical , therapeutic use : Adrenocorticotropic Hormone, Antibodies, Monoclonal, Immunoglobulins, Immunologic Factors.
- adverse effects : Antineoplastic Combined Chemotherapy Protocols.
- drug effects : B-Lymphocytes.
- drug therapy : Ataxia, Myoclonus.
- immunology : B-Lymphocytes.
- methods : Drug Therapy, Combination, Lymphocyte Depletion.
- pathology : B-Lymphocytes.
- physiopathology : Opsoclonus-Myoclonus Syndrome.
- therapeutic use : Antineoplastic Combined Chemotherapy Protocols.
- therapy : Opsoclonus-Myoclonus Syndrome.
- Analysis of Variance, Antibodies, Monoclonal, Murine-Derived, Child, Preschool, Drug Administration Schedule, Female, Humans, Infant, Male, Treatment Outcome.
Abstract
Twelve immunotherapy‐naïve children with opsoclonus‐myoclonus syndrome and CSF B cell expansion received rituximab, adrenocorticotropic hormone (ACTH), and IVIg. Motor severity lessened 73% by 6 mo and 81% at 1 yr (P < 0.0001). Opsoclonus and action myoclonus disappeared rapidly, whereas gait ataxia and some other motor components improved more slowly. ACTH dose was tapered by 87%. Reduction in total CSF B cells was profound at 6 mo (‐93%). By study end, peripheral B cells returned to 53% of baseline and serum IgM levels to 63%. Overall clinical response trailed peripheral B cell and IgM depletion, but improvement continued after their levels recovered. All but one non‐ambulatory subject became ambulatory without additional chemotherapy; two relapsed and remitted; four had rituximab‐related or possibly related adverse events; and two had low‐titer human anti‐chimeric antibody. Combination of rituximab with conventional agents as initial therapy was effective and safe. A controlled trial with long‐term safety monitoring is indicated. © 2009 Movement Disorder Society
Url:
DOI: 10.1002/mds.22941
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Disord.</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><date type="published" when="2010-01-30">2010-01-30</date><biblScope unit="vol">25</biblScope><biblScope unit="issue">2</biblScope><biblScope unit="page" from="238">238</biblScope><biblScope unit="page" to="242">242</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">492CFB8EF5800D55950B99390DFDF727D300BDC6</idno><idno type="DOI">10.1002/mds.22941</idno><idno type="ArticleID">MDS22941</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>ACTH</term><term>Adrenocorticotropic Hormone (adverse effects)</term><term>Adrenocorticotropic Hormone (therapeutic use)</term><term>Analysis of Variance</term><term>Antibodies, Monoclonal (adverse effects)</term><term>Antibodies, Monoclonal (therapeutic use)</term><term>Antibodies, Monoclonal, Murine-Derived</term><term>Antineoplastic Combined Chemotherapy Protocols (adverse effects)</term><term>Antineoplastic Combined Chemotherapy Protocols (therapeutic use)</term><term>Ataxia (drug therapy)</term><term>B-Lymphocytes (drug effects)</term><term>B-Lymphocytes (immunology)</term><term>B-Lymphocytes (pathology)</term><term>Child, Preschool</term><term>Drug Administration Schedule</term><term>Drug Therapy, Combination (methods)</term><term>Female</term><term>Humans</term><term>Immunoglobulin M (blood)</term><term>Immunoglobulins (adverse effects)</term><term>Immunoglobulins (therapeutic use)</term><term>Immunologic Factors (adverse effects)</term><term>Immunologic Factors (therapeutic use)</term><term>Infant</term><term>Kinsbourne syndrome</term><term>Lymphocyte Depletion (methods)</term><term>Male</term><term>Myoclonus (drug therapy)</term><term>Opsoclonus-Myoclonus Syndrome (physiopathology)</term><term>Opsoclonus-Myoclonus Syndrome (therapy)</term><term>Treatment Outcome</term><term>anti‐B cell agent</term><term>dancing eyes</term><term>neuroblastoma</term><term>paraneoplastic syndrome</term><term>rituximab</term></keywords><keywords scheme="MESH" type="chemical" qualifier="adverse effects" xml:lang="en"><term>Adrenocorticotropic Hormone</term><term>Antibodies, Monoclonal</term><term>Immunoglobulins</term><term>Immunologic Factors</term></keywords><keywords scheme="MESH" type="chemical" qualifier="blood" xml:lang="en"><term>Immunoglobulin M</term></keywords><keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Adrenocorticotropic Hormone</term><term>Antibodies, Monoclonal</term><term>Immunoglobulins</term><term>Immunologic Factors</term></keywords><keywords scheme="MESH" qualifier="adverse effects" xml:lang="en"><term>Antineoplastic Combined Chemotherapy Protocols</term></keywords><keywords scheme="MESH" qualifier="drug effects" xml:lang="en"><term>B-Lymphocytes</term></keywords><keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Ataxia</term><term>Myoclonus</term></keywords><keywords scheme="MESH" qualifier="immunology" xml:lang="en"><term>B-Lymphocytes</term></keywords><keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Drug Therapy, Combination</term><term>Lymphocyte Depletion</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>B-Lymphocytes</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Opsoclonus-Myoclonus Syndrome</term></keywords><keywords scheme="MESH" qualifier="therapeutic use" xml:lang="en"><term>Antineoplastic Combined Chemotherapy Protocols</term></keywords><keywords scheme="MESH" qualifier="therapy" xml:lang="en"><term>Opsoclonus-Myoclonus Syndrome</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Analysis of Variance</term><term>Antibodies, Monoclonal, Murine-Derived</term><term>Child, Preschool</term><term>Drug Administration Schedule</term><term>Female</term><term>Humans</term><term>Infant</term><term>Male</term><term>Treatment Outcome</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Twelve immunotherapy‐naïve children with opsoclonus‐myoclonus syndrome and CSF B cell expansion received rituximab, adrenocorticotropic hormone (ACTH), and IVIg. Motor severity lessened 73% by 6 mo and 81% at 1 yr (P < 0.0001). Opsoclonus and action myoclonus disappeared rapidly, whereas gait ataxia and some other motor components improved more slowly. ACTH dose was tapered by 87%. Reduction in total CSF B cells was profound at 6 mo (‐93%). By study end, peripheral B cells returned to 53% of baseline and serum IgM levels to 63%. Overall clinical response trailed peripheral B cell and IgM depletion, but improvement continued after their levels recovered. All but one non‐ambulatory subject became ambulatory without additional chemotherapy; two relapsed and remitted; four had rituximab‐related or possibly related adverse events; and two had low‐titer human anti‐chimeric antibody. Combination of rituximab with conventional agents as initial therapy was effective and safe. A controlled trial with long‐term safety monitoring is indicated. © 2009 Movement Disorder Society</div></front></TEI><double doi="10.1002/mds.22941"><ISTEX><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">B cell depletion therapy for new‐onset opsoclonus‐myoclonus</title><author><name sortKey="Pranzatelli, Michael R" sort="Pranzatelli, Michael R" uniqKey="Pranzatelli M" first="Michael R." last="Pranzatelli">Michael R. Pranzatelli</name></author><author><name sortKey="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D." last="Tate">Elizabeth D. Tate</name></author><author><name sortKey="Swan, Jennifer A" sort="Swan, Jennifer A" uniqKey="Swan J" first="Jennifer A." last="Swan">Jennifer A. Swan</name></author><author><name sortKey="Travelstead, Anna L" sort="Travelstead, Anna L" uniqKey="Travelstead A" first="Anna L." last="Travelstead">Anna L. Travelstead</name></author><author><name sortKey="Colliver, Jerry A" sort="Colliver, Jerry A" uniqKey="Colliver J" first="Jerry A." last="Colliver">Jerry A. Colliver</name></author><author><name sortKey="Verhulst, Steven J" sort="Verhulst, Steven J" uniqKey="Verhulst S" first="Steven J." last="Verhulst">Steven J. Verhulst</name></author><author><name sortKey="Crosley, Carl J" sort="Crosley, Carl J" uniqKey="Crosley C" first="Carl J." last="Crosley">Carl J. Crosley</name></author><author><name sortKey="Graf, William D" sort="Graf, William D" uniqKey="Graf W" first="William D." last="Graf">William D. Graf</name></author><author><name sortKey="Joseph, Suja A" sort="Joseph, Suja A" uniqKey="Joseph S" first="Suja A." last="Joseph">Suja A. Joseph</name></author><author><name sortKey="Kelfer, Howard M" sort="Kelfer, Howard M" uniqKey="Kelfer H" first="Howard M." last="Kelfer">Howard M. Kelfer</name></author><author><name sortKey="Raju, G Praveen" sort="Raju, G Praveen" uniqKey="Raju G" first="G. Praveen" last="Raju">G. Praveen Raju</name></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:492CFB8EF5800D55950B99390DFDF727D300BDC6</idno><date when="2010" year="2010">2010</date><idno type="doi">10.1002/mds.22941</idno><idno type="url">https://api.istex.fr/document/492CFB8EF5800D55950B99390DFDF727D300BDC6/fulltext/pdf</idno><idno type="wicri:Area/Istex/Corpus">000C26</idno><idno type="wicri:Area/Istex/Curation">000C26</idno><idno type="wicri:Area/Istex/Checkpoint">000B19</idno><idno type="wicri:doubleKey">0885-3185:2010:Pranzatelli M:b:cell:depletion</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">B cell depletion therapy for new‐onset opsoclonus‐myoclonus</title><author><name sortKey="Pranzatelli, Michael R" sort="Pranzatelli, Michael R" uniqKey="Pranzatelli M" first="Michael R." last="Pranzatelli">Michael R. Pranzatelli</name><affiliation><wicri:noCountry code="no comma">National Pediatric Myoclonus Center and Departments of Neurology</wicri:noCountry></affiliation></author><author><name sortKey="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D." last="Tate">Elizabeth D. Tate</name><affiliation><wicri:noCountry code="no comma">National Pediatric Myoclonus Center and Departments of Neurology</wicri:noCountry></affiliation></author><author><name sortKey="Swan, Jennifer A" sort="Swan, Jennifer A" uniqKey="Swan J" first="Jennifer A." last="Swan">Jennifer A. Swan</name><affiliation><wicri:noCountry code="no comma">National Pediatric Myoclonus Center and Departments of Neurology</wicri:noCountry></affiliation></author><author><name sortKey="Travelstead, Anna L" sort="Travelstead, Anna L" uniqKey="Travelstead A" first="Anna L." last="Travelstead">Anna L. Travelstead</name><affiliation><wicri:noCountry code="no comma">Flow Cytometry Facility</wicri:noCountry></affiliation></author><author><name sortKey="Colliver, Jerry A" sort="Colliver, Jerry A" uniqKey="Colliver J" first="Jerry A." last="Colliver">Jerry A. Colliver</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Biostatistics and Research Consulting, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></affiliation></author><author><name sortKey="Verhulst, Steven J" sort="Verhulst, Steven J" uniqKey="Verhulst S" first="Steven J." last="Verhulst">Steven J. Verhulst</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Biostatistics and Research Consulting, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></affiliation></author><author><name sortKey="Crosley, Carl J" sort="Crosley, Carl J" uniqKey="Crosley C" first="Carl J." last="Crosley">Carl J. Crosley</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, SUNY, Syracuse, New York</wicri:regionArea><placeName><region type="state">État de New York</region></placeName></affiliation></author><author><name sortKey="Graf, William D" sort="Graf, William D" uniqKey="Graf W" first="William D." last="Graf">William D. Graf</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Section of Child Neurology, Children's Mercy Hospitals and Clinics, Kansas City, Missouri</wicri:regionArea><placeName><region type="state">Missouri (État)</region></placeName></affiliation></author><author><name sortKey="Joseph, Suja A" sort="Joseph, Suja A" uniqKey="Joseph S" first="Suja A." last="Joseph">Suja A. Joseph</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Medical College of Wisconsin, Milwaukee, Wisconsin</wicri:regionArea><placeName><region type="state">Wisconsin</region></placeName></affiliation></author><author><name sortKey="Kelfer, Howard M" sort="Kelfer, Howard M" uniqKey="Kelfer H" first="Howard M." last="Kelfer">Howard M. Kelfer</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Cook Children's Medical Center, Fort Worth, Texas</wicri:regionArea><placeName><region type="state">Texas</region></placeName></affiliation></author><author><name sortKey="Raju, G Praveen" sort="Raju, G Praveen" uniqKey="Raju G" first="G. Praveen" last="Raju">G. Praveen Raju</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Pediatrics, Harvard Medical School, Children's Hospital Boston, Boston, Massachusetts</wicri:regionArea><placeName><region type="state">Massachusetts</region></placeName></affiliation></author></analytic><monogr></monogr><series><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. Disord.</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><date type="published" when="2010-01-30">2010-01-30</date><biblScope unit="vol">25</biblScope><biblScope unit="issue">2</biblScope><biblScope unit="page" from="238">238</biblScope><biblScope unit="page" to="242">242</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">492CFB8EF5800D55950B99390DFDF727D300BDC6</idno><idno type="DOI">10.1002/mds.22941</idno><idno type="ArticleID">MDS22941</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>ACTH</term><term>Kinsbourne syndrome</term><term>anti‐B cell agent</term><term>dancing eyes</term><term>neuroblastoma</term><term>paraneoplastic syndrome</term><term>rituximab</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Twelve immunotherapy‐naïve children with opsoclonus‐myoclonus syndrome and CSF B cell expansion received rituximab, adrenocorticotropic hormone (ACTH), and IVIg. Motor severity lessened 73% by 6 mo and 81% at 1 yr (P < 0.0001). Opsoclonus and action myoclonus disappeared rapidly, whereas gait ataxia and some other motor components improved more slowly. ACTH dose was tapered by 87%. Reduction in total CSF B cells was profound at 6 mo (‐93%). By study end, peripheral B cells returned to 53% of baseline and serum IgM levels to 63%. Overall clinical response trailed peripheral B cell and IgM depletion, but improvement continued after their levels recovered. All but one non‐ambulatory subject became ambulatory without additional chemotherapy; two relapsed and remitted; four had rituximab‐related or possibly related adverse events; and two had low‐titer human anti‐chimeric antibody. Combination of rituximab with conventional agents as initial therapy was effective and safe. A controlled trial with long‐term safety monitoring is indicated. © 2009 Movement Disorder Society</div></front></TEI></ISTEX><PubMed><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">B cell depletion therapy for new-onset opsoclonus-myoclonus.</title><author><name sortKey="Pranzatelli, Michael R" sort="Pranzatelli, Michael R" uniqKey="Pranzatelli M" first="Michael R" last="Pranzatelli">Michael R. Pranzatelli</name><affiliation wicri:level="1"><nlm:affiliation>National Pediatric Myoclonus Center, Southern Illinois University School of Medicine, Springfield, Illinois 62794-9643, USA. mpranzatelli@siumed.edu</nlm:affiliation><country xml:lang="fr">États-Unis</country><wicri:regionArea>National Pediatric Myoclonus Center, Southern Illinois University School of Medicine, Springfield, Illinois 62794-9643</wicri:regionArea><wicri:noRegion>Illinois 62794-9643</wicri:noRegion></affiliation></author><author><name sortKey="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D" last="Tate">Elizabeth D. Tate</name></author><author><name sortKey="Swan, Jennifer A" sort="Swan, Jennifer A" uniqKey="Swan J" first="Jennifer A" last="Swan">Jennifer A. Swan</name></author><author><name sortKey="Travelstead, Anna L" sort="Travelstead, Anna L" uniqKey="Travelstead A" first="Anna L" last="Travelstead">Anna L. Travelstead</name></author><author><name sortKey="Colliver, Jerry A" sort="Colliver, Jerry A" uniqKey="Colliver J" first="Jerry A" last="Colliver">Jerry A. Colliver</name></author><author><name sortKey="Verhulst, Steven J" sort="Verhulst, Steven J" uniqKey="Verhulst S" first="Steven J" last="Verhulst">Steven J. Verhulst</name></author><author><name sortKey="Crosley, Carl J" sort="Crosley, Carl J" uniqKey="Crosley C" first="Carl J" last="Crosley">Carl J. Crosley</name></author><author><name sortKey="Graf, William D" sort="Graf, William D" uniqKey="Graf W" first="William D" last="Graf">William D. Graf</name></author><author><name sortKey="Joseph, Suja A" sort="Joseph, Suja A" uniqKey="Joseph S" first="Suja A" last="Joseph">Suja A. Joseph</name></author><author><name sortKey="Kelfer, Howard M" sort="Kelfer, Howard M" uniqKey="Kelfer H" first="Howard M" last="Kelfer">Howard M. Kelfer</name></author><author><name sortKey="Raju, G Praveen" sort="Raju, G Praveen" uniqKey="Raju G" first="G Praveen" last="Raju">G Praveen Raju</name></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2010">2010</date><idno type="doi">10.1002/mds.22941</idno><idno type="RBID">pubmed:20063398</idno><idno type="pmid">20063398</idno><idno type="wicri:Area/PubMed/Corpus">001A43</idno><idno type="wicri:Area/PubMed/Curation">001A43</idno><idno type="wicri:Area/PubMed/Checkpoint">001A30</idno><idno type="wicri:Area/Ncbi/Merge">002959</idno><idno type="wicri:Area/Ncbi/Curation">002959</idno><idno type="wicri:Area/Ncbi/Checkpoint">002959</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">B cell depletion therapy for new-onset opsoclonus-myoclonus.</title><author><name sortKey="Pranzatelli, Michael R" sort="Pranzatelli, Michael R" uniqKey="Pranzatelli M" first="Michael R" last="Pranzatelli">Michael R. Pranzatelli</name><affiliation wicri:level="1"><nlm:affiliation>National Pediatric Myoclonus Center, Southern Illinois University School of Medicine, Springfield, Illinois 62794-9643, USA. mpranzatelli@siumed.edu</nlm:affiliation><country xml:lang="fr">États-Unis</country><wicri:regionArea>National Pediatric Myoclonus Center, Southern Illinois University School of Medicine, Springfield, Illinois 62794-9643</wicri:regionArea><wicri:noRegion>Illinois 62794-9643</wicri:noRegion></affiliation></author><author><name sortKey="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D" last="Tate">Elizabeth D. Tate</name></author><author><name sortKey="Swan, Jennifer A" sort="Swan, Jennifer A" uniqKey="Swan J" first="Jennifer A" last="Swan">Jennifer A. Swan</name></author><author><name sortKey="Travelstead, Anna L" sort="Travelstead, Anna L" uniqKey="Travelstead A" first="Anna L" last="Travelstead">Anna L. Travelstead</name></author><author><name sortKey="Colliver, Jerry A" sort="Colliver, Jerry A" uniqKey="Colliver J" first="Jerry A" last="Colliver">Jerry A. Colliver</name></author><author><name sortKey="Verhulst, Steven J" sort="Verhulst, Steven J" uniqKey="Verhulst S" first="Steven J" last="Verhulst">Steven J. Verhulst</name></author><author><name sortKey="Crosley, Carl J" sort="Crosley, Carl J" uniqKey="Crosley C" first="Carl J" last="Crosley">Carl J. Crosley</name></author><author><name sortKey="Graf, William D" sort="Graf, William D" uniqKey="Graf W" first="William D" last="Graf">William D. Graf</name></author><author><name sortKey="Joseph, Suja A" sort="Joseph, Suja A" uniqKey="Joseph S" first="Suja A" last="Joseph">Suja A. Joseph</name></author><author><name sortKey="Kelfer, Howard M" sort="Kelfer, Howard M" uniqKey="Kelfer H" first="Howard M" last="Kelfer">Howard M. Kelfer</name></author><author><name sortKey="Raju, G Praveen" sort="Raju, G Praveen" uniqKey="Raju G" first="G Praveen" last="Raju">G Praveen Raju</name></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="eISSN">1531-8257</idno><imprint><date when="2010" type="published">2010</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adrenocorticotropic Hormone (adverse effects)</term><term>Adrenocorticotropic Hormone (therapeutic use)</term><term>Analysis of Variance</term><term>Antibodies, Monoclonal (adverse effects)</term><term>Antibodies, Monoclonal (therapeutic use)</term><term>Antibodies, Monoclonal, Murine-Derived</term><term>Antineoplastic Combined Chemotherapy Protocols (adverse effects)</term><term>Antineoplastic Combined Chemotherapy Protocols (therapeutic use)</term><term>Ataxia (drug therapy)</term><term>B-Lymphocytes (drug effects)</term><term>B-Lymphocytes (immunology)</term><term>B-Lymphocytes (pathology)</term><term>Child, Preschool</term><term>Drug Administration Schedule</term><term>Drug Therapy, Combination (methods)</term><term>Female</term><term>Humans</term><term>Immunoglobulin M (blood)</term><term>Immunoglobulins (adverse effects)</term><term>Immunoglobulins (therapeutic use)</term><term>Immunologic Factors (adverse effects)</term><term>Immunologic Factors (therapeutic use)</term><term>Infant</term><term>Lymphocyte Depletion (methods)</term><term>Male</term><term>Myoclonus (drug therapy)</term><term>Opsoclonus-Myoclonus Syndrome (physiopathology)</term><term>Opsoclonus-Myoclonus Syndrome (therapy)</term><term>Treatment Outcome</term></keywords><keywords scheme="MESH" type="chemical" qualifier="adverse effects" xml:lang="en"><term>Adrenocorticotropic Hormone</term><term>Antibodies, Monoclonal</term><term>Immunoglobulins</term><term>Immunologic Factors</term></keywords><keywords scheme="MESH" type="chemical" qualifier="blood" xml:lang="en"><term>Immunoglobulin M</term></keywords><keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Adrenocorticotropic Hormone</term><term>Antibodies, Monoclonal</term><term>Immunoglobulins</term><term>Immunologic Factors</term></keywords><keywords scheme="MESH" qualifier="adverse effects" xml:lang="en"><term>Antineoplastic Combined Chemotherapy Protocols</term></keywords><keywords scheme="MESH" qualifier="drug effects" xml:lang="en"><term>B-Lymphocytes</term></keywords><keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Ataxia</term><term>Myoclonus</term></keywords><keywords scheme="MESH" qualifier="immunology" xml:lang="en"><term>B-Lymphocytes</term></keywords><keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Drug Therapy, Combination</term><term>Lymphocyte Depletion</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>B-Lymphocytes</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Opsoclonus-Myoclonus Syndrome</term></keywords><keywords scheme="MESH" qualifier="therapeutic use" xml:lang="en"><term>Antineoplastic Combined Chemotherapy Protocols</term></keywords><keywords scheme="MESH" qualifier="therapy" xml:lang="en"><term>Opsoclonus-Myoclonus Syndrome</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Analysis of Variance</term><term>Antibodies, Monoclonal, Murine-Derived</term><term>Child, Preschool</term><term>Drug Administration Schedule</term><term>Female</term><term>Humans</term><term>Infant</term><term>Male</term><term>Treatment Outcome</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Twelve immunotherapy-naïve children with opsoclonus-myoclonus syndrome and CSF B cell expansion received rituximab, adrenocorticotropic hormone (ACTH), and IVIg. Motor severity lessened 73% by 6 mo and 81% at 1 yr (P < 0.0001). Opsoclonus and action myoclonus disappeared rapidly, whereas gait ataxia and some other motor components improved more slowly. ACTH dose was tapered by 87%. Reduction in total CSF B cells was profound at 6 mo (-93%). By study end, peripheral B cells returned to 53% of baseline and serum IgM levels to 63%. Overall clinical response trailed peripheral B cell and IgM depletion, but improvement continued after their levels recovered. All but one non-ambulatory subject became ambulatory without additional chemotherapy; two relapsed and remitted; four had rituximab-related or possibly related adverse events; and two had low-titer human anti-chimeric antibody. Combination of rituximab with conventional agents as initial therapy was effective and safe. A controlled trial with long-term safety monitoring is indicated.</div></front></TEI></PubMed></double></record>Pour manipuler ce document sous Unix (Dilib)
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