CSF B‐cell expansion in opsoclonus‐myoclonus syndrome: A biomarker of disease activity
Identifieur interne : 005748 ( Main/Merge ); précédent : 005747; suivant : 005749CSF B‐cell expansion in opsoclonus‐myoclonus syndrome: A biomarker of disease activity
Auteurs : Michael R. Pranzatelli [États-Unis] ; Anna L. Travelstead [États-Unis] ; Elizabeth D. Tate [États-Unis] ; Tyler J. Allison [États-Unis] ; Steven J. Verhulst [États-Unis]Source :
- Movement Disorders [ 0885-3185 ] ; 2004-07.
English descriptors
- KwdEn :
- Adolescent, Antigens, CD19 (cerebrospinal fluid), Antigens, CD19 (immunology), Antigens, CD5 (cerebrospinal fluid), Antigens, CD5 (immunology), B lymphocytes, B-Lymphocytes, Biological Markers, Brain Neoplasms (complications), B‐cell trafficking, Child, Child, Preschool, Female, Flow Cytometry, Humans, Immunophenotyping, Infant, Kinsbourne syndrome, Lymphocyte Subsets, Male, Neuroblastoma (complications), Paraneoplastic Syndromes, Nervous System (cerebrospinal fluid), Paraneoplastic Syndromes, Nervous System (complications), Paraneoplastic Syndromes, Nervous System (immunology), Severity of Illness Index, Videotape Recording, myoclonic disorders, neuroblastoma, paraneoplastic syndrome.
- MESH :
- chemical , cerebrospinal fluid : Antigens, CD19, Antigens, CD5.
- chemical , immunology : Antigens, CD19, Antigens, CD5.
- cerebrospinal fluid : Paraneoplastic Syndromes, Nervous System.
- complications : Brain Neoplasms, Neuroblastoma, Paraneoplastic Syndromes, Nervous System.
- immunology : Paraneoplastic Syndromes, Nervous System.
- Adolescent, B-Lymphocytes, Biological Markers, Child, Child, Preschool, Female, Flow Cytometry, Humans, Immunophenotyping, Infant, Lymphocyte Subsets, Male, Severity of Illness Index, Videotape Recording.
Abstract
Lack of a biomarker of disease activity has hindered the therapy of childhood opsoclonus‐myoclonus syndrome (OMS), which is purported to be mediated humorally. To determine if the cerebrospinal fluid (CSF) B lymphocyte, which may traffic into the central nervous system (CNS) to produce antibody locally, is one such biomarker, B lymphocytes were immunophenotyped in the CSF and blood of 56 children with OMS and 26 pediatric controls by dual‐laser flow cytometry. Neurological severity was rated blindly from videotapes using a validated 12‐item motor evaluation scale. Children with OMS manifested a 4‐ to 7‐fold higher percentage of total B‐cells in CSF (P < 0.0001), including CD5+ (P = 0.001) and CD5− (P = 0.0004) B‐cell subsets, compared with controls, in whom the percentages were negligible and unchanging with age. CSF expansion of both B‐cell subsets increased with disease severity and decreased with disease duration (P ≤ 0.0001, ANOVA). Previous treatment with conventional immunotherapies, chemotherapy, or tumor resection had not normalized B‐cell percentages in those with lingering symptoms. These studies reveal that CSF B‐cell expansion in OMS is characteristic and often persistent. Presence of the autoreactive CD5+ B‐cell subset and correlations with neurological severity and disease duration suggest CSF B‐cell expansion is a biomarker of disease activity and possible target for B‐cell–specific therapy. Immunophenotyping of CSF lymphocytes by flow cytometry yields valuable clinical information missed by routine studies and allows crucial treatment decisions to be made rapidly. © 2004 Movement Disorder Society
Url:
DOI: 10.1002/mds.20125
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ISTEX:8C425EAC22CC04F03FB3BDD5483A51470CE6F751Le document en format XML
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To determine if the cerebrospinal fluid (CSF) B lymphocyte, which may traffic into the central nervous system (CNS) to produce antibody locally, is one such biomarker, B lymphocytes were immunophenotyped in the CSF and blood of 56 children with OMS and 26 pediatric controls by dual‐laser flow cytometry. Neurological severity was rated blindly from videotapes using a validated 12‐item motor evaluation scale. Children with OMS manifested a 4‐ to 7‐fold higher percentage of total B‐cells in CSF (P < 0.0001), including CD5+ (P = 0.001) and CD5− (P = 0.0004) B‐cell subsets, compared with controls, in whom the percentages were negligible and unchanging with age. CSF expansion of both B‐cell subsets increased with disease severity and decreased with disease duration (P ≤ 0.0001, ANOVA). Previous treatment with conventional immunotherapies, chemotherapy, or tumor resection had not normalized B‐cell percentages in those with lingering symptoms. These studies reveal that CSF B‐cell expansion in OMS is characteristic and often persistent. Presence of the autoreactive CD5+ B‐cell subset and correlations with neurological severity and disease duration suggest CSF B‐cell expansion is a biomarker of disease activity and possible target for B‐cell–specific therapy. Immunophenotyping of CSF lymphocytes by flow cytometry yields valuable clinical information missed by routine studies and allows crucial treatment decisions to be made rapidly. © 2004 Movement Disorder Society</div></front></TEI><double doi="10.1002/mds.20125"><ISTEX><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">CSF B‐cell expansion in opsoclonus‐myoclonus syndrome: A biomarker of disease activity</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="Travelstead, Anna L" sort="Travelstead, Anna L" uniqKey="Travelstead A" first="Anna L." last="Travelstead">Anna L. Travelstead</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="Allison, Tyler J" sort="Allison, Tyler J" uniqKey="Allison T" first="Tyler J." last="Allison">Tyler J. Allison</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></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:8C425EAC22CC04F03FB3BDD5483A51470CE6F751</idno><date when="2004" year="2004">2004</date><idno type="doi">10.1002/mds.20125</idno><idno type="url">https://api.istex.fr/document/8C425EAC22CC04F03FB3BDD5483A51470CE6F751/fulltext/pdf</idno><idno type="wicri:Area/Istex/Corpus">000760</idno><idno type="wicri:Area/Istex/Curation">000760</idno><idno type="wicri:Area/Istex/Checkpoint">002771</idno><idno type="wicri:doubleKey">0885-3185:2004:Pranzatelli M:csf:b:cell</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">CSF B‐cell expansion in opsoclonus‐myoclonus syndrome: A biomarker of disease activity</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="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></affiliation><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Pediatrics, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></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:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Medical Microbiology and Immunology, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></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:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></affiliation></author><author><name sortKey="Allison, Tyler J" sort="Allison, Tyler J" uniqKey="Allison T" first="Tyler J." last="Allison">Tyler J. Allison</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, 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>Statistics and Research Consulting, Southern Illinois University School of Medicine, Springfield, Illinois</wicri:regionArea><placeName><region type="state">Illinois</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="2004-07">2004-07</date><biblScope unit="vol">19</biblScope><biblScope unit="issue">7</biblScope><biblScope unit="page" from="770">770</biblScope><biblScope unit="page" to="777">777</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">8C425EAC22CC04F03FB3BDD5483A51470CE6F751</idno><idno type="DOI">10.1002/mds.20125</idno><idno type="ArticleID">MDS20125</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>B lymphocytes</term><term>B‐cell trafficking</term><term>Kinsbourne syndrome</term><term>myoclonic disorders</term><term>neuroblastoma</term><term>paraneoplastic syndrome</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Lack of a biomarker of disease activity has hindered the therapy of childhood opsoclonus‐myoclonus syndrome (OMS), which is purported to be mediated humorally. To determine if the cerebrospinal fluid (CSF) B lymphocyte, which may traffic into the central nervous system (CNS) to produce antibody locally, is one such biomarker, B lymphocytes were immunophenotyped in the CSF and blood of 56 children with OMS and 26 pediatric controls by dual‐laser flow cytometry. Neurological severity was rated blindly from videotapes using a validated 12‐item motor evaluation scale. Children with OMS manifested a 4‐ to 7‐fold higher percentage of total B‐cells in CSF (P < 0.0001), including CD5+ (P = 0.001) and CD5− (P = 0.0004) B‐cell subsets, compared with controls, in whom the percentages were negligible and unchanging with age. CSF expansion of both B‐cell subsets increased with disease severity and decreased with disease duration (P ≤ 0.0001, ANOVA). Previous treatment with conventional immunotherapies, chemotherapy, or tumor resection had not normalized B‐cell percentages in those with lingering symptoms. These studies reveal that CSF B‐cell expansion in OMS is characteristic and often persistent. Presence of the autoreactive CD5+ B‐cell subset and correlations with neurological severity and disease duration suggest CSF B‐cell expansion is a biomarker of disease activity and possible target for B‐cell–specific therapy. Immunophenotyping of CSF lymphocytes by flow cytometry yields valuable clinical information missed by routine studies and allows crucial treatment decisions to be made rapidly. © 2004 Movement Disorder Society</div></front></TEI></ISTEX><PubMed><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">CSF B-cell expansion in opsoclonus-myoclonus syndrome: a biomarker of disease activity.</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>Department of Neurology, 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>Department of Neurology, Southern Illinois University School of Medicine, Springfield, Illinois 62794-9643</wicri:regionArea><wicri:noRegion>Illinois 62794-9643</wicri:noRegion></affiliation></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="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D" last="Tate">Elizabeth D. Tate</name></author><author><name sortKey="Allison, Tyler J" sort="Allison, Tyler J" uniqKey="Allison T" first="Tyler J" last="Allison">Tyler J. Allison</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></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2004">2004</date><idno type="RBID">pubmed:15254934</idno><idno type="pmid">15254934</idno><idno type="doi">10.1002/mds.20125</idno><idno type="wicri:Area/PubMed/Corpus">003418</idno><idno type="wicri:Area/PubMed/Curation">003418</idno><idno type="wicri:Area/PubMed/Checkpoint">003538</idno><idno type="wicri:Area/Ncbi/Merge">000E66</idno><idno type="wicri:Area/Ncbi/Curation">000E66</idno><idno type="wicri:Area/Ncbi/Checkpoint">000E66</idno><idno type="wicri:doubleKey">0885-3185:2004:Pranzatelli M:csf:b:cell</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">CSF B-cell expansion in opsoclonus-myoclonus syndrome: a biomarker of disease activity.</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>Department of Neurology, 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>Department of Neurology, Southern Illinois University School of Medicine, Springfield, Illinois 62794-9643</wicri:regionArea><wicri:noRegion>Illinois 62794-9643</wicri:noRegion></affiliation></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="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D" last="Tate">Elizabeth D. Tate</name></author><author><name sortKey="Allison, Tyler J" sort="Allison, Tyler J" uniqKey="Allison T" first="Tyler J" last="Allison">Tyler J. Allison</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></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="ISSN">0885-3185</idno><imprint><date when="2004" type="published">2004</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term><term>Antigens, CD19 (cerebrospinal fluid)</term><term>Antigens, CD19 (immunology)</term><term>Antigens, CD5 (cerebrospinal fluid)</term><term>Antigens, CD5 (immunology)</term><term>B-Lymphocytes</term><term>Biological Markers</term><term>Brain Neoplasms (complications)</term><term>Child</term><term>Child, Preschool</term><term>Female</term><term>Flow Cytometry</term><term>Humans</term><term>Immunophenotyping</term><term>Infant</term><term>Lymphocyte Subsets</term><term>Male</term><term>Neuroblastoma (complications)</term><term>Paraneoplastic Syndromes, Nervous System (cerebrospinal fluid)</term><term>Paraneoplastic Syndromes, Nervous System (complications)</term><term>Paraneoplastic Syndromes, Nervous System (immunology)</term><term>Severity of Illness Index</term><term>Videotape Recording</term></keywords><keywords scheme="MESH" type="chemical" qualifier="cerebrospinal fluid" xml:lang="en"><term>Antigens, CD19</term><term>Antigens, CD5</term></keywords><keywords scheme="MESH" type="chemical" qualifier="immunology" xml:lang="en"><term>Antigens, CD19</term><term>Antigens, CD5</term></keywords><keywords scheme="MESH" qualifier="cerebrospinal fluid" xml:lang="en"><term>Paraneoplastic Syndromes, Nervous System</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Brain Neoplasms</term><term>Neuroblastoma</term><term>Paraneoplastic Syndromes, Nervous System</term></keywords><keywords scheme="MESH" qualifier="immunology" xml:lang="en"><term>Paraneoplastic Syndromes, Nervous System</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>B-Lymphocytes</term><term>Biological Markers</term><term>Child</term><term>Child, Preschool</term><term>Female</term><term>Flow Cytometry</term><term>Humans</term><term>Immunophenotyping</term><term>Infant</term><term>Lymphocyte Subsets</term><term>Male</term><term>Severity of Illness Index</term><term>Videotape Recording</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Lack of a biomarker of disease activity has hindered the therapy of childhood opsoclonus-myoclonus syndrome (OMS), which is purported to be mediated humorally. To determine if the cerebrospinal fluid (CSF) B lymphocyte, which may traffic into the central nervous system (CNS) to produce antibody locally, is one such biomarker, B lymphocytes were immunophenotyped in the CSF and blood of 56 children with OMS and 26 pediatric controls by dual-laser flow cytometry. Neurological severity was rated blindly from videotapes using a validated 12-item motor evaluation scale. Children with OMS manifested a 4- to 7-fold higher percentage of total B-cells in CSF (P < 0.0001), including CD5(+) (P = 0.001) and CD5(-) (P = 0.0004) B-cell subsets, compared with controls, in whom the percentages were negligible and unchanging with age. CSF expansion of both B-cell subsets increased with disease severity and decreased with disease duration (P </div></front></TEI></PubMed></double></record>Pour manipuler ce document sous Unix (Dilib)
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