Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica
Background: Recently we showed that antibodies to myelin oligodendrocyte glycoprotein (MOG) can be found in aquaporin-4 (AQP4)-immunoglobulin (IgG) seronegative pediatric and adult patients with definite and high-risk neuromyelitis optica (NMO). Objective: The purpose of this study was to describe t...
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| Veröffentlicht in: | Multiple sclerosis 2013-07, Vol.19 (8), p.1052-1059 |
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| creator | Rostásy, K Mader, S Hennes, EM Schanda, K Gredler, V Guenther, A Blaschek, A Korenke, C Pritsch, M Pohl, D Maier, O Kuchukhidze, G Brunner-Krainz, M Berger, T Reindl, M |
| description | Background:
Recently we showed that antibodies to myelin oligodendrocyte glycoprotein (MOG) can be found in aquaporin-4 (AQP4)-immunoglobulin (IgG) seronegative pediatric and adult patients with definite and high-risk neuromyelitis optica (NMO).
Objective:
The purpose of this study was to describe the clinical characteristics and temporal dynamics of MOG-IgG in AQP4-IgG seronegative pediatric patients presenting with definite NMO.
Methods:
Children with definite NMO who were referred for further testing of serum antibodies for AQP4 and MOG with a cell-based assay were included in this study. Clinical disease course, cerebrospinal fluid and magnetic resonance imaging (MRI) studies of these patients were reviewed.
Results:
Between 2008 and 2012 eight children who fulfilled the diagnostic criteria of definite NMO were recruited. Two children with definite NMO tested positive for AQP4-IgG but were negative for MOG-IgG antibodies. Three children had an absence of AQP4-IgG and MOG-IgG antibodies. Three children with definite NMO had high titers of serum MOG-IgG antibodies (≥1: 160), but no AQP4-directed humoral immune response. Longitudinal analysis of serum samples of the latter three children showed persisting high MOG-IgG titers over time.
Conclusion:
Pediatric patients presenting with clinical symptoms and MRI findings highly suggestive of NMO but with high and persisting MOG-IgG antibody titers are most likely to represent a distinct subgroup of acute demyelinating diseases with important clinical and therapeutic implications. |
| doi_str_mv | 10.1177/1352458512470310 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_1381100288</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sage_id>10.1177_1352458512470310</sage_id><sourcerecordid>1381100288</sourcerecordid><originalsourceid>FETCH-LOGICAL-c461t-1a62ffde38dc421776ef7c7c62e3aed736c18c2eed8b9981e4964956b7b55a0c3</originalsourceid><addsrcrecordid>eNp1kc1rFTEUxYMo9kP3rmRABDejuUkmmVlKUSsU2kVdD5nkzpAyL5kmGWH-e_P6XlUKXeXj_O6593IIeQf0M4BSX4A3TDRtA0woyoG-IKcglKppp-jLci9yvddPyFlKd5RSpXjzmpwwzholGZyS7QZjcik7P1W7DWfnqzC7KVj0NgazZaymeTNhiSFjEbXPbgjWYar2r_tVLyE6X4tHZas8Tjq731gtaJ3O0ZnytcbwYJ9dqsKSndFvyKtRzwnfHs9z8uv7t9uLy_rq-sfPi69XtREScg1asnG0yFtrBCtLSxyVUUYy5Bqt4tJAaxiibYeuawFFJ0XXyEENTaOp4efk08G3rHC_Ysr9ziWD86w9hjX1wFsASlnbFvTDE_QurNGX6QpVOglgShaKHigTQ0oRx36Jbqfj1gPt97H0T2MpJe-PxuuwQ_u34DGHAnw8AjoZPY9Re-PSP04VQ_rA1Qcu6Qn_m-65xn8AEZKj8g</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1373641276</pqid></control><display><type>article</type><title>Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica</title><source>SAGE Complete A-Z List</source><source>MEDLINE</source><creator>Rostásy, K ; Mader, S ; Hennes, EM ; Schanda, K ; Gredler, V ; Guenther, A ; Blaschek, A ; Korenke, C ; Pritsch, M ; Pohl, D ; Maier, O ; Kuchukhidze, G ; Brunner-Krainz, M ; Berger, T ; Reindl, M</creator><creatorcontrib>Rostásy, K ; Mader, S ; Hennes, EM ; Schanda, K ; Gredler, V ; Guenther, A ; Blaschek, A ; Korenke, C ; Pritsch, M ; Pohl, D ; Maier, O ; Kuchukhidze, G ; Brunner-Krainz, M ; Berger, T ; Reindl, M</creatorcontrib><description>Background:
Recently we showed that antibodies to myelin oligodendrocyte glycoprotein (MOG) can be found in aquaporin-4 (AQP4)-immunoglobulin (IgG) seronegative pediatric and adult patients with definite and high-risk neuromyelitis optica (NMO).
Objective:
The purpose of this study was to describe the clinical characteristics and temporal dynamics of MOG-IgG in AQP4-IgG seronegative pediatric patients presenting with definite NMO.
Methods:
Children with definite NMO who were referred for further testing of serum antibodies for AQP4 and MOG with a cell-based assay were included in this study. Clinical disease course, cerebrospinal fluid and magnetic resonance imaging (MRI) studies of these patients were reviewed.
Results:
Between 2008 and 2012 eight children who fulfilled the diagnostic criteria of definite NMO were recruited. Two children with definite NMO tested positive for AQP4-IgG but were negative for MOG-IgG antibodies. Three children had an absence of AQP4-IgG and MOG-IgG antibodies. Three children with definite NMO had high titers of serum MOG-IgG antibodies (≥1: 160), but no AQP4-directed humoral immune response. Longitudinal analysis of serum samples of the latter three children showed persisting high MOG-IgG titers over time.
Conclusion:
Pediatric patients presenting with clinical symptoms and MRI findings highly suggestive of NMO but with high and persisting MOG-IgG antibody titers are most likely to represent a distinct subgroup of acute demyelinating diseases with important clinical and therapeutic implications.</description><identifier>ISSN: 1352-4585</identifier><identifier>EISSN: 1477-0970</identifier><identifier>DOI: 10.1177/1352458512470310</identifier><identifier>PMID: 23257621</identifier><language>eng</language><publisher>London, England: SAGE Publications</publisher><subject>Adolescent ; Adult ; Aquaporin 4 - immunology ; Autoantibodies - blood ; Autoantibodies - immunology ; Autoantigens - immunology ; Biological and medical sciences ; Child ; Degenerative and inherited degenerative diseases of the nervous system. Leukodystrophies. Prion diseases ; Female ; Humans ; Immunoglobulin G - immunology ; Magnetic Resonance Imaging ; Male ; Medical sciences ; Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis ; Myelin-Oligodendrocyte Glycoprotein - immunology ; Neurology ; Neuromyelitis Optica - blood ; Neuromyelitis Optica - immunology</subject><ispartof>Multiple sclerosis, 2013-07, Vol.19 (8), p.1052-1059</ispartof><rights>The Author(s) 2012</rights><rights>2014 INIST-CNRS</rights><rights>SAGE Publications © Jul 2013</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c461t-1a62ffde38dc421776ef7c7c62e3aed736c18c2eed8b9981e4964956b7b55a0c3</citedby><cites>FETCH-LOGICAL-c461t-1a62ffde38dc421776ef7c7c62e3aed736c18c2eed8b9981e4964956b7b55a0c3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://journals.sagepub.com/doi/pdf/10.1177/1352458512470310$$EPDF$$P50$$Gsage$$H</linktopdf><linktohtml>$$Uhttps://journals.sagepub.com/doi/10.1177/1352458512470310$$EHTML$$P50$$Gsage$$H</linktohtml><link.rule.ids>314,776,780,21798,27901,27902,43597,43598</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=27524021$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/23257621$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Rostásy, K</creatorcontrib><creatorcontrib>Mader, S</creatorcontrib><creatorcontrib>Hennes, EM</creatorcontrib><creatorcontrib>Schanda, K</creatorcontrib><creatorcontrib>Gredler, V</creatorcontrib><creatorcontrib>Guenther, A</creatorcontrib><creatorcontrib>Blaschek, A</creatorcontrib><creatorcontrib>Korenke, C</creatorcontrib><creatorcontrib>Pritsch, M</creatorcontrib><creatorcontrib>Pohl, D</creatorcontrib><creatorcontrib>Maier, O</creatorcontrib><creatorcontrib>Kuchukhidze, G</creatorcontrib><creatorcontrib>Brunner-Krainz, M</creatorcontrib><creatorcontrib>Berger, T</creatorcontrib><creatorcontrib>Reindl, M</creatorcontrib><title>Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica</title><title>Multiple sclerosis</title><addtitle>Mult Scler</addtitle><description>Background:
Recently we showed that antibodies to myelin oligodendrocyte glycoprotein (MOG) can be found in aquaporin-4 (AQP4)-immunoglobulin (IgG) seronegative pediatric and adult patients with definite and high-risk neuromyelitis optica (NMO).
Objective:
The purpose of this study was to describe the clinical characteristics and temporal dynamics of MOG-IgG in AQP4-IgG seronegative pediatric patients presenting with definite NMO.
Methods:
Children with definite NMO who were referred for further testing of serum antibodies for AQP4 and MOG with a cell-based assay were included in this study. Clinical disease course, cerebrospinal fluid and magnetic resonance imaging (MRI) studies of these patients were reviewed.
Results:
Between 2008 and 2012 eight children who fulfilled the diagnostic criteria of definite NMO were recruited. Two children with definite NMO tested positive for AQP4-IgG but were negative for MOG-IgG antibodies. Three children had an absence of AQP4-IgG and MOG-IgG antibodies. Three children with definite NMO had high titers of serum MOG-IgG antibodies (≥1: 160), but no AQP4-directed humoral immune response. Longitudinal analysis of serum samples of the latter three children showed persisting high MOG-IgG titers over time.
Conclusion:
Pediatric patients presenting with clinical symptoms and MRI findings highly suggestive of NMO but with high and persisting MOG-IgG antibody titers are most likely to represent a distinct subgroup of acute demyelinating diseases with important clinical and therapeutic implications.</description><subject>Adolescent</subject><subject>Adult</subject><subject>Aquaporin 4 - immunology</subject><subject>Autoantibodies - blood</subject><subject>Autoantibodies - immunology</subject><subject>Autoantigens - immunology</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Degenerative and inherited degenerative diseases of the nervous system. Leukodystrophies. Prion diseases</subject><subject>Female</subject><subject>Humans</subject><subject>Immunoglobulin G - immunology</subject><subject>Magnetic Resonance Imaging</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis</subject><subject>Myelin-Oligodendrocyte Glycoprotein - immunology</subject><subject>Neurology</subject><subject>Neuromyelitis Optica - blood</subject><subject>Neuromyelitis Optica - immunology</subject><issn>1352-4585</issn><issn>1477-0970</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><recordid>eNp1kc1rFTEUxYMo9kP3rmRABDejuUkmmVlKUSsU2kVdD5nkzpAyL5kmGWH-e_P6XlUKXeXj_O6593IIeQf0M4BSX4A3TDRtA0woyoG-IKcglKppp-jLci9yvddPyFlKd5RSpXjzmpwwzholGZyS7QZjcik7P1W7DWfnqzC7KVj0NgazZaymeTNhiSFjEbXPbgjWYar2r_tVLyE6X4tHZas8Tjq731gtaJ3O0ZnytcbwYJ9dqsKSndFvyKtRzwnfHs9z8uv7t9uLy_rq-sfPi69XtREScg1asnG0yFtrBCtLSxyVUUYy5Bqt4tJAaxiibYeuawFFJ0XXyEENTaOp4efk08G3rHC_Ysr9ziWD86w9hjX1wFsASlnbFvTDE_QurNGX6QpVOglgShaKHigTQ0oRx36Jbqfj1gPt97H0T2MpJe-PxuuwQ_u34DGHAnw8AjoZPY9Re-PSP04VQ_rA1Qcu6Qn_m-65xn8AEZKj8g</recordid><startdate>20130701</startdate><enddate>20130701</enddate><creator>Rostásy, K</creator><creator>Mader, S</creator><creator>Hennes, EM</creator><creator>Schanda, K</creator><creator>Gredler, V</creator><creator>Guenther, A</creator><creator>Blaschek, A</creator><creator>Korenke, C</creator><creator>Pritsch, M</creator><creator>Pohl, D</creator><creator>Maier, O</creator><creator>Kuchukhidze, G</creator><creator>Brunner-Krainz, M</creator><creator>Berger, T</creator><creator>Reindl, M</creator><general>SAGE Publications</general><general>Sage Publications</general><general>Sage Publications Ltd</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7T5</scope><scope>7TK</scope><scope>7U9</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>H94</scope><scope>K9-</scope><scope>K9.</scope><scope>M0R</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope></search><sort><creationdate>20130701</creationdate><title>Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica</title><author>Rostásy, K ; Mader, S ; Hennes, EM ; Schanda, K ; Gredler, V ; Guenther, A ; Blaschek, A ; Korenke, C ; Pritsch, M ; Pohl, D ; Maier, O ; Kuchukhidze, G ; Brunner-Krainz, M ; Berger, T ; Reindl, M</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c461t-1a62ffde38dc421776ef7c7c62e3aed736c18c2eed8b9981e4964956b7b55a0c3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2013</creationdate><topic>Adolescent</topic><topic>Adult</topic><topic>Aquaporin 4 - immunology</topic><topic>Autoantibodies - blood</topic><topic>Autoantibodies - immunology</topic><topic>Autoantigens - immunology</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Degenerative and inherited degenerative diseases of the nervous system. Leukodystrophies. Prion diseases</topic><topic>Female</topic><topic>Humans</topic><topic>Immunoglobulin G - immunology</topic><topic>Magnetic Resonance Imaging</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis</topic><topic>Myelin-Oligodendrocyte Glycoprotein - immunology</topic><topic>Neurology</topic><topic>Neuromyelitis Optica - blood</topic><topic>Neuromyelitis Optica - immunology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Rostásy, K</creatorcontrib><creatorcontrib>Mader, S</creatorcontrib><creatorcontrib>Hennes, EM</creatorcontrib><creatorcontrib>Schanda, K</creatorcontrib><creatorcontrib>Gredler, V</creatorcontrib><creatorcontrib>Guenther, A</creatorcontrib><creatorcontrib>Blaschek, A</creatorcontrib><creatorcontrib>Korenke, C</creatorcontrib><creatorcontrib>Pritsch, M</creatorcontrib><creatorcontrib>Pohl, D</creatorcontrib><creatorcontrib>Maier, O</creatorcontrib><creatorcontrib>Kuchukhidze, G</creatorcontrib><creatorcontrib>Brunner-Krainz, M</creatorcontrib><creatorcontrib>Berger, T</creatorcontrib><creatorcontrib>Reindl, M</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>Virology and AIDS Abstracts</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>Consumer Health Database (Alumni Edition)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Consumer Health Database</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><jtitle>Multiple sclerosis</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Rostásy, K</au><au>Mader, S</au><au>Hennes, EM</au><au>Schanda, K</au><au>Gredler, V</au><au>Guenther, A</au><au>Blaschek, A</au><au>Korenke, C</au><au>Pritsch, M</au><au>Pohl, D</au><au>Maier, O</au><au>Kuchukhidze, G</au><au>Brunner-Krainz, M</au><au>Berger, T</au><au>Reindl, M</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica</atitle><jtitle>Multiple sclerosis</jtitle><addtitle>Mult Scler</addtitle><date>2013-07-01</date><risdate>2013</risdate><volume>19</volume><issue>8</issue><spage>1052</spage><epage>1059</epage><pages>1052-1059</pages><issn>1352-4585</issn><eissn>1477-0970</eissn><abstract>Background:
Recently we showed that antibodies to myelin oligodendrocyte glycoprotein (MOG) can be found in aquaporin-4 (AQP4)-immunoglobulin (IgG) seronegative pediatric and adult patients with definite and high-risk neuromyelitis optica (NMO).
Objective:
The purpose of this study was to describe the clinical characteristics and temporal dynamics of MOG-IgG in AQP4-IgG seronegative pediatric patients presenting with definite NMO.
Methods:
Children with definite NMO who were referred for further testing of serum antibodies for AQP4 and MOG with a cell-based assay were included in this study. Clinical disease course, cerebrospinal fluid and magnetic resonance imaging (MRI) studies of these patients were reviewed.
Results:
Between 2008 and 2012 eight children who fulfilled the diagnostic criteria of definite NMO were recruited. Two children with definite NMO tested positive for AQP4-IgG but were negative for MOG-IgG antibodies. Three children had an absence of AQP4-IgG and MOG-IgG antibodies. Three children with definite NMO had high titers of serum MOG-IgG antibodies (≥1: 160), but no AQP4-directed humoral immune response. Longitudinal analysis of serum samples of the latter three children showed persisting high MOG-IgG titers over time.
Conclusion:
Pediatric patients presenting with clinical symptoms and MRI findings highly suggestive of NMO but with high and persisting MOG-IgG antibody titers are most likely to represent a distinct subgroup of acute demyelinating diseases with important clinical and therapeutic implications.</abstract><cop>London, England</cop><pub>SAGE Publications</pub><pmid>23257621</pmid><doi>10.1177/1352458512470310</doi><tpages>8</tpages></addata></record> |
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| ispartof | Multiple sclerosis, 2013-07, Vol.19 (8), p.1052-1059 |
| issn | 1352-4585 1477-0970 |
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| source | SAGE Complete A-Z List; MEDLINE |
| subjects | Adolescent Adult Aquaporin 4 - immunology Autoantibodies - blood Autoantibodies - immunology Autoantigens - immunology Biological and medical sciences Child Degenerative and inherited degenerative diseases of the nervous system. Leukodystrophies. Prion diseases Female Humans Immunoglobulin G - immunology Magnetic Resonance Imaging Male Medical sciences Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis Myelin-Oligodendrocyte Glycoprotein - immunology Neurology Neuromyelitis Optica - blood Neuromyelitis Optica - immunology |
| title | Persisting myelin oligodendrocyte glycoprotein antibodies in aquaporin-4 antibody negative pediatric neuromyelitis optica |
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