CSF B-cell expansion in opsoclonus-myoclonus syndrome: A biomarker of disease activity
Identifieur interne : 002222 ( PascalFrancis/Checkpoint ); précédent : 002221; suivant : 002223CSF 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.
Descripteurs français
- Pascal (Inist)
English descriptors
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.
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Travelstead</name><affiliation wicri:level="2"><inist:fA14 i1="03"><s1>Department of Medical Microbiology and Immunology, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>2 aut.</sZ></inist:fA14><country>États-Unis</country><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"><inist:fA14 i1="01"><s1>Department of Neurology, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>1 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ></inist:fA14><country>États-Unis</country><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. 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Verhulst</name><affiliation wicri:level="2"><inist:fA14 i1="04"><s1>Statitics and Research Consulting, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>5 aut.</sZ></inist:fA14><country>États-Unis</country><placeName><region type="state">Illinois</region></placeName></affiliation></author></analytic><series><title level="j" type="main">Movement disorders</title><title level="j" type="abbreviated">Mov. disord.</title><idno type="ISSN">0885-3185</idno><imprint><date when="2004">2004</date></imprint></series></biblStruct></sourceDesc><seriesStmt><title level="j" type="main">Movement disorders</title><title level="j" type="abbreviated">Mov. disord.</title><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Cerebrospinal fluid</term><term>Myoclonus</term><term>Nervous system diseases</term><term>Opsoclonus</term><term>β Cell</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Opsoclonie</term><term>Liquide céphalorachidien</term><term>Cellule β</term><term>Myoclonie</term><term>Système nerveux pathologie</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<sup>+</sup> (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<sup>+</sup> 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.</div></front></TEI><inist><standard h6="B"><pA><fA01 i1="01" i2="1"><s0>0885-3185</s0></fA01><fA03 i2="1"><s0>Mov. disord.</s0></fA03><fA05><s2>19</s2></fA05><fA06><s2>7</s2></fA06><fA08 i1="01" i2="1" l="ENG"><s1>CSF B-cell expansion in opsoclonus-myoclonus syndrome: A biomarker of disease activity</s1></fA08><fA11 i1="01" i2="1"><s1>PRANZATELLI (Michael R.)</s1></fA11><fA11 i1="02" i2="1"><s1>TRAVELSTEAD (Anna L.)</s1></fA11><fA11 i1="03" i2="1"><s1>TATE (Elizabeth D.)</s1></fA11><fA11 i1="04" i2="1"><s1>ALLISON (Tyler J.)</s1></fA11><fA11 i1="05" i2="1"><s1>VERHULST (Steven J.)</s1></fA11><fA14 i1="01"><s1>Department of Neurology, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>1 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ></fA14><fA14 i1="02"><s1>Department of Pediatrics, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>1 aut.</sZ></fA14><fA14 i1="03"><s1>Department of Medical Microbiology and Immunology, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>2 aut.</sZ></fA14><fA14 i1="04"><s1>Statitics and Research Consulting, Southern Illinois University School of Medicine</s1><s2>Springfield, Illinois</s2><s3>USA</s3><sZ>5 aut.</sZ></fA14><fA20><s1>770-777</s1></fA20><fA21><s1>2004</s1></fA21><fA23 i1="01"><s0>ENG</s0></fA23><fA43 i1="01"><s1>INIST</s1><s2>20953</s2><s5>354000113929420040</s5></fA43><fA44><s0>0000</s0><s1>© 2004 INIST-CNRS. 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Pranzatelli</name></region><name sortKey="Allison, Tyler J" sort="Allison, Tyler J" uniqKey="Allison T" first="Tyler J." last="Allison">Tyler J. Allison</name><name sortKey="Pranzatelli, Michael R" sort="Pranzatelli, Michael R" uniqKey="Pranzatelli M" first="Michael R." last="Pranzatelli">Michael R. Pranzatelli</name><name sortKey="Tate, Elizabeth D" sort="Tate, Elizabeth D" uniqKey="Tate E" first="Elizabeth D." last="Tate">Elizabeth D. Tate</name><name sortKey="Travelstead, Anna L" sort="Travelstead, Anna L" uniqKey="Travelstead A" first="Anna L." last="Travelstead">Anna L. Travelstead</name><name sortKey="Verhulst, Steven J" sort="Verhulst, Steven J" uniqKey="Verhulst S" first="Steven J." last="Verhulst">Steven J. Verhulst</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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