Long-term clinical, MRI, and cognitive follow-up in a large cohort of pathologically confirmed, predominantly tumefactive multiple sclerosis
Background: Limited studies have described long-term outcomes in pathology confirmed multiple sclerosis (MS). Objectives: To describe long-term clinical–radiographic–cognitive outcomes in a prospectively followed cohort of patients with pathologically confirmed CNS demyelinating disease, consistent...
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| Veröffentlicht in: | Multiple sclerosis 2022-03, Vol.28 (3), p.441-452 |
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| creator | Kalinowska-Lyszczarz, Alicja Tillema, Jan-Mendelt Tobin, W Oliver Guo, Yong Fitz-Gibbon, Patrick D Weigand, Stephen D Giraldo-Chica, Monica Port, John D Lucchinetti, Claudia F |
| description | Background:
Limited studies have described long-term outcomes in pathology confirmed multiple sclerosis (MS).
Objectives:
To describe long-term clinical–radiographic–cognitive outcomes in a prospectively followed cohort of patients with pathologically confirmed CNS demyelinating disease, consistent with MS.
Methods:
Subjects underwent clinical assessment, standardized 3T-MRI brain, and cognitive battery.
Results:
Seventy-five patients were included. Biopsied lesion size was ⩾ 2 cm in 62/75. At follow-up, median duration since biopsy was 11 years. Median EDSS was 3 and lesion burden was large (median 10 cm3). At follow-up, 57/75 met MS criteria, 17/75 had clinically isolated syndrome, and 1 radiographic changes only. Disability scores were comparable to a prevalence cohort in Olmsted County (p < 0.001, n = 218). Cognitive outcomes below age-normed standards included psychomotor, attention, working memory, and executive function domains. Total lesion volume and index lesion-related severity correlated with EDSS and cognitive performance. Volumetric cortical/subcortical GM correlated less than lesion metrics to cognitive outcomes.
Conclusion:
Despite early aggressive course in pathologically confirmed MS, its long-term course was comparable to typical MS in our study. Cognitive impairment in this group seemed to correlate strongest to index lesion severity and total lesion volume. It remains to be established how the aggressive nature of the lesion, biopsy, and treatment affect clinical/cognitive outcomes. |
| doi_str_mv | 10.1177/13524585211024162 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2548415286</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sage_id>10.1177_13524585211024162</sage_id><sourcerecordid>2635448643</sourcerecordid><originalsourceid>FETCH-LOGICAL-c368t-275810e293bc6bb2d7e187e25cd3c01ef93a3f159fedc3fe07ca581726028213</originalsourceid><addsrcrecordid>eNp1kd1qFTEUhYNU-mcfwBsJ9MaLMzW_kzmXpdRaOCJI74eczM40JZNMk4zSd-hDm-OpCopXCaxvreydhdBbSi4oVeoD5ZIJ2UlGKWGCtuwVOqZCqYasFTmo96o3O-AIneT8QAhRistDdMQFo0xJeYyeNzGMTYE0YeNdcEb7Ff789XaFdRiwiWNwxX0DbKP38XuzzNgFrLHXaYQq38dUcLR41uU--jju_P6pCsG6NMGwwnOCIU4u6FCqUJYJrDY_I6fFFzd7wNl4SDG7_Aa9ttpnOHs5T9Hdx-u7q0_N5svN7dXlpjG87UpTJ-8oAbbmW9Nut2xQQDsFTJqBG0LBrrnmlsq1hcFwC0QZXR2KtYR1jPJT9H4fO6f4uEAu_eSyAe91gLjknknRCSpZ11b0_C_0IS4p1OF61nIpRNcKXim6p0xdIyew_ZzcpNNTT0m_a6r_p6nqefeSvGzrR_12_KqmAhd7IOsR_jz7_8Qf20CcWg</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2635448643</pqid></control><display><type>article</type><title>Long-term clinical, MRI, and cognitive follow-up in a large cohort of pathologically confirmed, predominantly tumefactive multiple sclerosis</title><source>Access via SAGE</source><source>MEDLINE</source><creator>Kalinowska-Lyszczarz, Alicja ; Tillema, Jan-Mendelt ; Tobin, W Oliver ; Guo, Yong ; Fitz-Gibbon, Patrick D ; Weigand, Stephen D ; Giraldo-Chica, Monica ; Port, John D ; Lucchinetti, Claudia F</creator><creatorcontrib>Kalinowska-Lyszczarz, Alicja ; Tillema, Jan-Mendelt ; Tobin, W Oliver ; Guo, Yong ; Fitz-Gibbon, Patrick D ; Weigand, Stephen D ; Giraldo-Chica, Monica ; Port, John D ; Lucchinetti, Claudia F</creatorcontrib><description>Background:
Limited studies have described long-term outcomes in pathology confirmed multiple sclerosis (MS).
Objectives:
To describe long-term clinical–radiographic–cognitive outcomes in a prospectively followed cohort of patients with pathologically confirmed CNS demyelinating disease, consistent with MS.
Methods:
Subjects underwent clinical assessment, standardized 3T-MRI brain, and cognitive battery.
Results:
Seventy-five patients were included. Biopsied lesion size was ⩾ 2 cm in 62/75. At follow-up, median duration since biopsy was 11 years. Median EDSS was 3 and lesion burden was large (median 10 cm3). At follow-up, 57/75 met MS criteria, 17/75 had clinically isolated syndrome, and 1 radiographic changes only. Disability scores were comparable to a prevalence cohort in Olmsted County (p < 0.001, n = 218). Cognitive outcomes below age-normed standards included psychomotor, attention, working memory, and executive function domains. Total lesion volume and index lesion-related severity correlated with EDSS and cognitive performance. Volumetric cortical/subcortical GM correlated less than lesion metrics to cognitive outcomes.
Conclusion:
Despite early aggressive course in pathologically confirmed MS, its long-term course was comparable to typical MS in our study. Cognitive impairment in this group seemed to correlate strongest to index lesion severity and total lesion volume. It remains to be established how the aggressive nature of the lesion, biopsy, and treatment affect clinical/cognitive outcomes.</description><identifier>ISSN: 1352-4585</identifier><identifier>EISSN: 1477-0970</identifier><identifier>DOI: 10.1177/13524585211024162</identifier><identifier>PMID: 34212755</identifier><language>eng</language><publisher>London, England: SAGE Publications</publisher><subject>Age ; Biopsy ; Brain - pathology ; Cognition ; Cognitive ability ; Demyelinating diseases ; Demyelinating Diseases - diagnostic imaging ; Demyelinating Diseases - pathology ; Demyelination ; Executive function ; Follow-Up Studies ; Humans ; Lesions ; Magnetic Resonance Imaging ; Multiple Sclerosis ; Patients ; Short term memory</subject><ispartof>Multiple sclerosis, 2022-03, Vol.28 (3), p.441-452</ispartof><rights>The Author(s), 2021</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c368t-275810e293bc6bb2d7e187e25cd3c01ef93a3f159fedc3fe07ca581726028213</citedby><cites>FETCH-LOGICAL-c368t-275810e293bc6bb2d7e187e25cd3c01ef93a3f159fedc3fe07ca581726028213</cites><orcidid>0000-0002-8141-2394 ; 0000-0002-0618-1637</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://journals.sagepub.com/doi/pdf/10.1177/13524585211024162$$EPDF$$P50$$Gsage$$H</linktopdf><linktohtml>$$Uhttps://journals.sagepub.com/doi/10.1177/13524585211024162$$EHTML$$P50$$Gsage$$H</linktohtml><link.rule.ids>314,780,784,21819,27924,27925,43621,43622</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/34212755$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Kalinowska-Lyszczarz, Alicja</creatorcontrib><creatorcontrib>Tillema, Jan-Mendelt</creatorcontrib><creatorcontrib>Tobin, W Oliver</creatorcontrib><creatorcontrib>Guo, Yong</creatorcontrib><creatorcontrib>Fitz-Gibbon, Patrick D</creatorcontrib><creatorcontrib>Weigand, Stephen D</creatorcontrib><creatorcontrib>Giraldo-Chica, Monica</creatorcontrib><creatorcontrib>Port, John D</creatorcontrib><creatorcontrib>Lucchinetti, Claudia F</creatorcontrib><title>Long-term clinical, MRI, and cognitive follow-up in a large cohort of pathologically confirmed, predominantly tumefactive multiple sclerosis</title><title>Multiple sclerosis</title><addtitle>Mult Scler</addtitle><description>Background:
Limited studies have described long-term outcomes in pathology confirmed multiple sclerosis (MS).
Objectives:
To describe long-term clinical–radiographic–cognitive outcomes in a prospectively followed cohort of patients with pathologically confirmed CNS demyelinating disease, consistent with MS.
Methods:
Subjects underwent clinical assessment, standardized 3T-MRI brain, and cognitive battery.
Results:
Seventy-five patients were included. Biopsied lesion size was ⩾ 2 cm in 62/75. At follow-up, median duration since biopsy was 11 years. Median EDSS was 3 and lesion burden was large (median 10 cm3). At follow-up, 57/75 met MS criteria, 17/75 had clinically isolated syndrome, and 1 radiographic changes only. Disability scores were comparable to a prevalence cohort in Olmsted County (p < 0.001, n = 218). Cognitive outcomes below age-normed standards included psychomotor, attention, working memory, and executive function domains. Total lesion volume and index lesion-related severity correlated with EDSS and cognitive performance. Volumetric cortical/subcortical GM correlated less than lesion metrics to cognitive outcomes.
Conclusion:
Despite early aggressive course in pathologically confirmed MS, its long-term course was comparable to typical MS in our study. Cognitive impairment in this group seemed to correlate strongest to index lesion severity and total lesion volume. It remains to be established how the aggressive nature of the lesion, biopsy, and treatment affect clinical/cognitive outcomes.</description><subject>Age</subject><subject>Biopsy</subject><subject>Brain - pathology</subject><subject>Cognition</subject><subject>Cognitive ability</subject><subject>Demyelinating diseases</subject><subject>Demyelinating Diseases - diagnostic imaging</subject><subject>Demyelinating Diseases - pathology</subject><subject>Demyelination</subject><subject>Executive function</subject><subject>Follow-Up Studies</subject><subject>Humans</subject><subject>Lesions</subject><subject>Magnetic Resonance Imaging</subject><subject>Multiple Sclerosis</subject><subject>Patients</subject><subject>Short term memory</subject><issn>1352-4585</issn><issn>1477-0970</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp1kd1qFTEUhYNU-mcfwBsJ9MaLMzW_kzmXpdRaOCJI74eczM40JZNMk4zSd-hDm-OpCopXCaxvreydhdBbSi4oVeoD5ZIJ2UlGKWGCtuwVOqZCqYasFTmo96o3O-AIneT8QAhRistDdMQFo0xJeYyeNzGMTYE0YeNdcEb7Ff789XaFdRiwiWNwxX0DbKP38XuzzNgFrLHXaYQq38dUcLR41uU--jju_P6pCsG6NMGwwnOCIU4u6FCqUJYJrDY_I6fFFzd7wNl4SDG7_Aa9ttpnOHs5T9Hdx-u7q0_N5svN7dXlpjG87UpTJ-8oAbbmW9Nut2xQQDsFTJqBG0LBrrnmlsq1hcFwC0QZXR2KtYR1jPJT9H4fO6f4uEAu_eSyAe91gLjknknRCSpZ11b0_C_0IS4p1OF61nIpRNcKXim6p0xdIyew_ZzcpNNTT0m_a6r_p6nqefeSvGzrR_12_KqmAhd7IOsR_jz7_8Qf20CcWg</recordid><startdate>202203</startdate><enddate>202203</enddate><creator>Kalinowska-Lyszczarz, Alicja</creator><creator>Tillema, Jan-Mendelt</creator><creator>Tobin, W Oliver</creator><creator>Guo, Yong</creator><creator>Fitz-Gibbon, Patrick D</creator><creator>Weigand, Stephen D</creator><creator>Giraldo-Chica, Monica</creator><creator>Port, John D</creator><creator>Lucchinetti, Claudia F</creator><general>SAGE Publications</general><general>Sage Publications Ltd</general><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>7T5</scope><scope>7TK</scope><scope>7U9</scope><scope>H94</scope><scope>K9.</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-8141-2394</orcidid><orcidid>https://orcid.org/0000-0002-0618-1637</orcidid></search><sort><creationdate>202203</creationdate><title>Long-term clinical, MRI, and cognitive follow-up in a large cohort of pathologically confirmed, predominantly tumefactive multiple sclerosis</title><author>Kalinowska-Lyszczarz, Alicja ; Tillema, Jan-Mendelt ; Tobin, W Oliver ; Guo, Yong ; Fitz-Gibbon, Patrick D ; Weigand, Stephen D ; Giraldo-Chica, Monica ; Port, John D ; Lucchinetti, Claudia F</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c368t-275810e293bc6bb2d7e187e25cd3c01ef93a3f159fedc3fe07ca581726028213</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Age</topic><topic>Biopsy</topic><topic>Brain - pathology</topic><topic>Cognition</topic><topic>Cognitive ability</topic><topic>Demyelinating diseases</topic><topic>Demyelinating Diseases - diagnostic imaging</topic><topic>Demyelinating Diseases - pathology</topic><topic>Demyelination</topic><topic>Executive function</topic><topic>Follow-Up Studies</topic><topic>Humans</topic><topic>Lesions</topic><topic>Magnetic Resonance Imaging</topic><topic>Multiple Sclerosis</topic><topic>Patients</topic><topic>Short term memory</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Kalinowska-Lyszczarz, Alicja</creatorcontrib><creatorcontrib>Tillema, Jan-Mendelt</creatorcontrib><creatorcontrib>Tobin, W Oliver</creatorcontrib><creatorcontrib>Guo, Yong</creatorcontrib><creatorcontrib>Fitz-Gibbon, Patrick D</creatorcontrib><creatorcontrib>Weigand, Stephen D</creatorcontrib><creatorcontrib>Giraldo-Chica, Monica</creatorcontrib><creatorcontrib>Port, John D</creatorcontrib><creatorcontrib>Lucchinetti, Claudia F</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>Virology and AIDS Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>Multiple sclerosis</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Kalinowska-Lyszczarz, Alicja</au><au>Tillema, Jan-Mendelt</au><au>Tobin, W Oliver</au><au>Guo, Yong</au><au>Fitz-Gibbon, Patrick D</au><au>Weigand, Stephen D</au><au>Giraldo-Chica, Monica</au><au>Port, John D</au><au>Lucchinetti, Claudia F</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Long-term clinical, MRI, and cognitive follow-up in a large cohort of pathologically confirmed, predominantly tumefactive multiple sclerosis</atitle><jtitle>Multiple sclerosis</jtitle><addtitle>Mult Scler</addtitle><date>2022-03</date><risdate>2022</risdate><volume>28</volume><issue>3</issue><spage>441</spage><epage>452</epage><pages>441-452</pages><issn>1352-4585</issn><eissn>1477-0970</eissn><abstract>Background:
Limited studies have described long-term outcomes in pathology confirmed multiple sclerosis (MS).
Objectives:
To describe long-term clinical–radiographic–cognitive outcomes in a prospectively followed cohort of patients with pathologically confirmed CNS demyelinating disease, consistent with MS.
Methods:
Subjects underwent clinical assessment, standardized 3T-MRI brain, and cognitive battery.
Results:
Seventy-five patients were included. Biopsied lesion size was ⩾ 2 cm in 62/75. At follow-up, median duration since biopsy was 11 years. Median EDSS was 3 and lesion burden was large (median 10 cm3). At follow-up, 57/75 met MS criteria, 17/75 had clinically isolated syndrome, and 1 radiographic changes only. Disability scores were comparable to a prevalence cohort in Olmsted County (p < 0.001, n = 218). Cognitive outcomes below age-normed standards included psychomotor, attention, working memory, and executive function domains. Total lesion volume and index lesion-related severity correlated with EDSS and cognitive performance. Volumetric cortical/subcortical GM correlated less than lesion metrics to cognitive outcomes.
Conclusion:
Despite early aggressive course in pathologically confirmed MS, its long-term course was comparable to typical MS in our study. Cognitive impairment in this group seemed to correlate strongest to index lesion severity and total lesion volume. It remains to be established how the aggressive nature of the lesion, biopsy, and treatment affect clinical/cognitive outcomes.</abstract><cop>London, England</cop><pub>SAGE Publications</pub><pmid>34212755</pmid><doi>10.1177/13524585211024162</doi><tpages>12</tpages><orcidid>https://orcid.org/0000-0002-8141-2394</orcidid><orcidid>https://orcid.org/0000-0002-0618-1637</orcidid></addata></record> |
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| ispartof | Multiple sclerosis, 2022-03, Vol.28 (3), p.441-452 |
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| language | eng |
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| subjects | Age Biopsy Brain - pathology Cognition Cognitive ability Demyelinating diseases Demyelinating Diseases - diagnostic imaging Demyelinating Diseases - pathology Demyelination Executive function Follow-Up Studies Humans Lesions Magnetic Resonance Imaging Multiple Sclerosis Patients Short term memory |
| title | Long-term clinical, MRI, and cognitive follow-up in a large cohort of pathologically confirmed, predominantly tumefactive multiple sclerosis |
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