Motor, cognitive, and functional declines contribute to a single progressive factor in early HD
OBJECTIVE:To identify an improved measure of clinical progression in early Huntington disease (HD) using data from prospective observational cohort studies and placebo group data from randomized double-blind clinical trials. METHODS:We studied Unified Huntington Disease Rating Scale (UHDRS) and non-...
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| Veröffentlicht in: | Neurology 2017-12, Vol.89 (24), p.2495-2502 |
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| creator | Schobel, Scott A Palermo, Giuseppe Auinger, Peggy Long, Jeffrey D Ma, Shiyang Khwaja, Omar S Trundell, Dylan Cudkowicz, Merit Hersch, Steven Sampaio, Cristina Dorsey, E Ray Leavitt, Blair R Kieburtz, Karl D Sevigny, Jeffrey J Langbehn, Douglas R Tabrizi, Sarah J |
| description | OBJECTIVE:To identify an improved measure of clinical progression in early Huntington disease (HD) using data from prospective observational cohort studies and placebo group data from randomized double-blind clinical trials.
METHODS:We studied Unified Huntington Disease Rating Scale (UHDRS) and non-UHDRS clinical measures and brain measures of progressive atrophy in 1,668 individuals with early HD followed up prospectively for up to 30 to 36 months of longitudinal clinical follow-up.
RESULTS:The results demonstrated that a composite measure of motor, cognitive, and global functional decline best characterized clinical progression and was most strongly associated with brain measures of progressive corticostriatal atrophy.
CONCLUSIONS:Use of a composite motor, cognitive, and global functional clinical outcome measure in HD provides an improved measure of clinical progression more related to measures of progressive brain atrophy and provides an opportunity for enhanced clinical trial efficiency relative to currently used individual motor, cognitive, and functional outcome measures. |
| doi_str_mv | 10.1212/WNL.0000000000004743 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_5729794</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>1965247033</sourcerecordid><originalsourceid>FETCH-LOGICAL-c5083-fc43f92aa9970d48c40fc1247440b59ec829e68ca6dca9f37d61990be31790113</originalsourceid><addsrcrecordid>eNp9kUtPHDEQhC2UCBaSf4CQjzkwxK-ZsS-RIsIj0gYuiZKb5fX07Bq89sb2gPj3OFqCSA70xYf-qrqsQuiQkhPKKPv482p-Ql6M6AXfQTPasq7pOPv1Bs0IYbLhspd7aD_nG0Lqsle7aI8pKhiRaob0t1hiOsY2LoMr7g6OsQkDHqdgi4vBeDyA9S5ArkgoyS2mArhEbHB2YekBb1JcJsi5avFobHXDLmAwyT_gyy_v0NvR-Azvn94D9OP87PvpZTO_vvh6-nne2JZI3oxW8FExY5TqySCkFWS0lNU_CbJoFVjJFHTSmm6wRo28HzqqFFkAp70ilPID9Gnru5kWaxgs1KzG601ya5MedDRO_7sJbqWX8U63PVO9EtXgw5NBir8nyEWvXbbgvQkQp6yp6tqah3BeUbFFbYo5Jxifz1Ci_3Sjazf6_26q7OhlxGfR3zIqILfAffQFUr710z0kvQLjy-p170eoO5x-</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1965247033</pqid></control><display><type>article</type><title>Motor, cognitive, and functional declines contribute to a single progressive factor in early HD</title><source>MEDLINE</source><source>Alma/SFX Local Collection</source><source>Journals@Ovid Complete</source><creator>Schobel, Scott A ; Palermo, Giuseppe ; Auinger, Peggy ; Long, Jeffrey D ; Ma, Shiyang ; Khwaja, Omar S ; Trundell, Dylan ; Cudkowicz, Merit ; Hersch, Steven ; Sampaio, Cristina ; Dorsey, E Ray ; Leavitt, Blair R ; Kieburtz, Karl D ; Sevigny, Jeffrey J ; Langbehn, Douglas R ; Tabrizi, Sarah J</creator><creatorcontrib>Schobel, Scott A ; Palermo, Giuseppe ; Auinger, Peggy ; Long, Jeffrey D ; Ma, Shiyang ; Khwaja, Omar S ; Trundell, Dylan ; Cudkowicz, Merit ; Hersch, Steven ; Sampaio, Cristina ; Dorsey, E Ray ; Leavitt, Blair R ; Kieburtz, Karl D ; Sevigny, Jeffrey J ; Langbehn, Douglas R ; Tabrizi, Sarah J ; TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators ; For the TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</creatorcontrib><description>OBJECTIVE:To identify an improved measure of clinical progression in early Huntington disease (HD) using data from prospective observational cohort studies and placebo group data from randomized double-blind clinical trials.
METHODS:We studied Unified Huntington Disease Rating Scale (UHDRS) and non-UHDRS clinical measures and brain measures of progressive atrophy in 1,668 individuals with early HD followed up prospectively for up to 30 to 36 months of longitudinal clinical follow-up.
RESULTS:The results demonstrated that a composite measure of motor, cognitive, and global functional decline best characterized clinical progression and was most strongly associated with brain measures of progressive corticostriatal atrophy.
CONCLUSIONS:Use of a composite motor, cognitive, and global functional clinical outcome measure in HD provides an improved measure of clinical progression more related to measures of progressive brain atrophy and provides an opportunity for enhanced clinical trial efficiency relative to currently used individual motor, cognitive, and functional outcome measures.</description><identifier>ISSN: 0028-3878</identifier><identifier>EISSN: 1526-632X</identifier><identifier>DOI: 10.1212/WNL.0000000000004743</identifier><identifier>PMID: 29142089</identifier><language>eng</language><publisher>United States: American Academy of Neurology</publisher><subject>Adult ; Apathy ; Cognitive Dysfunction - physiopathology ; Cognitive Dysfunction - psychology ; Disease Progression ; Emotions ; Facial Recognition ; Female ; Humans ; Huntington Disease - drug therapy ; Huntington Disease - physiopathology ; Huntington Disease - psychology ; Longitudinal Studies ; Male ; Middle Aged ; Motor Skills ; Neuropsychological Tests ; Prospective Studies ; Randomized Controlled Trials as Topic ; Reproducibility of Results ; Signal-To-Noise Ratio ; Social Perception ; Stroop Test ; Ubiquinone - analogs & derivatives ; Ubiquinone - therapeutic use ; Vitamins - therapeutic use</subject><ispartof>Neurology, 2017-12, Vol.89 (24), p.2495-2502</ispartof><rights>2017 American Academy of Neurology</rights><rights>2017 American Academy of Neurology.</rights><rights>2017 American Academy of Neurology 2017 American Academy of Neurology</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c5083-fc43f92aa9970d48c40fc1247440b59ec829e68ca6dca9f37d61990be31790113</citedby><cites>FETCH-LOGICAL-c5083-fc43f92aa9970d48c40fc1247440b59ec829e68ca6dca9f37d61990be31790113</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,776,780,881,27901,27902</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/29142089$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Schobel, Scott A</creatorcontrib><creatorcontrib>Palermo, Giuseppe</creatorcontrib><creatorcontrib>Auinger, Peggy</creatorcontrib><creatorcontrib>Long, Jeffrey D</creatorcontrib><creatorcontrib>Ma, Shiyang</creatorcontrib><creatorcontrib>Khwaja, Omar S</creatorcontrib><creatorcontrib>Trundell, Dylan</creatorcontrib><creatorcontrib>Cudkowicz, Merit</creatorcontrib><creatorcontrib>Hersch, Steven</creatorcontrib><creatorcontrib>Sampaio, Cristina</creatorcontrib><creatorcontrib>Dorsey, E Ray</creatorcontrib><creatorcontrib>Leavitt, Blair R</creatorcontrib><creatorcontrib>Kieburtz, Karl D</creatorcontrib><creatorcontrib>Sevigny, Jeffrey J</creatorcontrib><creatorcontrib>Langbehn, Douglas R</creatorcontrib><creatorcontrib>Tabrizi, Sarah J</creatorcontrib><creatorcontrib>TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</creatorcontrib><creatorcontrib>For the TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</creatorcontrib><title>Motor, cognitive, and functional declines contribute to a single progressive factor in early HD</title><title>Neurology</title><addtitle>Neurology</addtitle><description>OBJECTIVE:To identify an improved measure of clinical progression in early Huntington disease (HD) using data from prospective observational cohort studies and placebo group data from randomized double-blind clinical trials.
METHODS:We studied Unified Huntington Disease Rating Scale (UHDRS) and non-UHDRS clinical measures and brain measures of progressive atrophy in 1,668 individuals with early HD followed up prospectively for up to 30 to 36 months of longitudinal clinical follow-up.
RESULTS:The results demonstrated that a composite measure of motor, cognitive, and global functional decline best characterized clinical progression and was most strongly associated with brain measures of progressive corticostriatal atrophy.
CONCLUSIONS:Use of a composite motor, cognitive, and global functional clinical outcome measure in HD provides an improved measure of clinical progression more related to measures of progressive brain atrophy and provides an opportunity for enhanced clinical trial efficiency relative to currently used individual motor, cognitive, and functional outcome measures.</description><subject>Adult</subject><subject>Apathy</subject><subject>Cognitive Dysfunction - physiopathology</subject><subject>Cognitive Dysfunction - psychology</subject><subject>Disease Progression</subject><subject>Emotions</subject><subject>Facial Recognition</subject><subject>Female</subject><subject>Humans</subject><subject>Huntington Disease - drug therapy</subject><subject>Huntington Disease - physiopathology</subject><subject>Huntington Disease - psychology</subject><subject>Longitudinal Studies</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Motor Skills</subject><subject>Neuropsychological Tests</subject><subject>Prospective Studies</subject><subject>Randomized Controlled Trials as Topic</subject><subject>Reproducibility of Results</subject><subject>Signal-To-Noise Ratio</subject><subject>Social Perception</subject><subject>Stroop Test</subject><subject>Ubiquinone - analogs & derivatives</subject><subject>Ubiquinone - therapeutic use</subject><subject>Vitamins - therapeutic use</subject><issn>0028-3878</issn><issn>1526-632X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kUtPHDEQhC2UCBaSf4CQjzkwxK-ZsS-RIsIj0gYuiZKb5fX07Bq89sb2gPj3OFqCSA70xYf-qrqsQuiQkhPKKPv482p-Ql6M6AXfQTPasq7pOPv1Bs0IYbLhspd7aD_nG0Lqsle7aI8pKhiRaob0t1hiOsY2LoMr7g6OsQkDHqdgi4vBeDyA9S5ArkgoyS2mArhEbHB2YekBb1JcJsi5avFobHXDLmAwyT_gyy_v0NvR-Azvn94D9OP87PvpZTO_vvh6-nne2JZI3oxW8FExY5TqySCkFWS0lNU_CbJoFVjJFHTSmm6wRo28HzqqFFkAp70ilPID9Gnru5kWaxgs1KzG601ya5MedDRO_7sJbqWX8U63PVO9EtXgw5NBir8nyEWvXbbgvQkQp6yp6tqah3BeUbFFbYo5Jxifz1Ci_3Sjazf6_26q7OhlxGfR3zIqILfAffQFUr710z0kvQLjy-p170eoO5x-</recordid><startdate>20171212</startdate><enddate>20171212</enddate><creator>Schobel, Scott A</creator><creator>Palermo, Giuseppe</creator><creator>Auinger, Peggy</creator><creator>Long, Jeffrey D</creator><creator>Ma, Shiyang</creator><creator>Khwaja, Omar S</creator><creator>Trundell, Dylan</creator><creator>Cudkowicz, Merit</creator><creator>Hersch, Steven</creator><creator>Sampaio, Cristina</creator><creator>Dorsey, E Ray</creator><creator>Leavitt, Blair R</creator><creator>Kieburtz, Karl D</creator><creator>Sevigny, Jeffrey J</creator><creator>Langbehn, Douglas R</creator><creator>Tabrizi, Sarah J</creator><general>American Academy of Neurology</general><general>Lippincott Williams & Wilkins</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>7X8</scope><scope>5PM</scope></search><sort><creationdate>20171212</creationdate><title>Motor, cognitive, and functional declines contribute to a single progressive factor in early HD</title><author>Schobel, Scott A ; Palermo, Giuseppe ; Auinger, Peggy ; Long, Jeffrey D ; Ma, Shiyang ; Khwaja, Omar S ; Trundell, Dylan ; Cudkowicz, Merit ; Hersch, Steven ; Sampaio, Cristina ; Dorsey, E Ray ; Leavitt, Blair R ; Kieburtz, Karl D ; Sevigny, Jeffrey J ; Langbehn, Douglas R ; Tabrizi, Sarah J</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c5083-fc43f92aa9970d48c40fc1247440b59ec829e68ca6dca9f37d61990be31790113</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Adult</topic><topic>Apathy</topic><topic>Cognitive Dysfunction - physiopathology</topic><topic>Cognitive Dysfunction - psychology</topic><topic>Disease Progression</topic><topic>Emotions</topic><topic>Facial Recognition</topic><topic>Female</topic><topic>Humans</topic><topic>Huntington Disease - drug therapy</topic><topic>Huntington Disease - physiopathology</topic><topic>Huntington Disease - psychology</topic><topic>Longitudinal Studies</topic><topic>Male</topic><topic>Middle Aged</topic><topic>Motor Skills</topic><topic>Neuropsychological Tests</topic><topic>Prospective Studies</topic><topic>Randomized Controlled Trials as Topic</topic><topic>Reproducibility of Results</topic><topic>Signal-To-Noise Ratio</topic><topic>Social Perception</topic><topic>Stroop Test</topic><topic>Ubiquinone - analogs & derivatives</topic><topic>Ubiquinone - therapeutic use</topic><topic>Vitamins - therapeutic use</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Schobel, Scott A</creatorcontrib><creatorcontrib>Palermo, Giuseppe</creatorcontrib><creatorcontrib>Auinger, Peggy</creatorcontrib><creatorcontrib>Long, Jeffrey D</creatorcontrib><creatorcontrib>Ma, Shiyang</creatorcontrib><creatorcontrib>Khwaja, Omar S</creatorcontrib><creatorcontrib>Trundell, Dylan</creatorcontrib><creatorcontrib>Cudkowicz, Merit</creatorcontrib><creatorcontrib>Hersch, Steven</creatorcontrib><creatorcontrib>Sampaio, Cristina</creatorcontrib><creatorcontrib>Dorsey, E Ray</creatorcontrib><creatorcontrib>Leavitt, Blair R</creatorcontrib><creatorcontrib>Kieburtz, Karl D</creatorcontrib><creatorcontrib>Sevigny, Jeffrey J</creatorcontrib><creatorcontrib>Langbehn, Douglas R</creatorcontrib><creatorcontrib>Tabrizi, Sarah J</creatorcontrib><creatorcontrib>TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</creatorcontrib><creatorcontrib>For the TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Schobel, Scott A</au><au>Palermo, Giuseppe</au><au>Auinger, Peggy</au><au>Long, Jeffrey D</au><au>Ma, Shiyang</au><au>Khwaja, Omar S</au><au>Trundell, Dylan</au><au>Cudkowicz, Merit</au><au>Hersch, Steven</au><au>Sampaio, Cristina</au><au>Dorsey, E Ray</au><au>Leavitt, Blair R</au><au>Kieburtz, Karl D</au><au>Sevigny, Jeffrey J</au><au>Langbehn, Douglas R</au><au>Tabrizi, Sarah J</au><aucorp>TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</aucorp><aucorp>For the TRACK-HD, COHORT, CARE-HD, and 2CARE Huntington Study Group Investigators</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Motor, cognitive, and functional declines contribute to a single progressive factor in early HD</atitle><jtitle>Neurology</jtitle><addtitle>Neurology</addtitle><date>2017-12-12</date><risdate>2017</risdate><volume>89</volume><issue>24</issue><spage>2495</spage><epage>2502</epage><pages>2495-2502</pages><issn>0028-3878</issn><eissn>1526-632X</eissn><abstract>OBJECTIVE:To identify an improved measure of clinical progression in early Huntington disease (HD) using data from prospective observational cohort studies and placebo group data from randomized double-blind clinical trials.
METHODS:We studied Unified Huntington Disease Rating Scale (UHDRS) and non-UHDRS clinical measures and brain measures of progressive atrophy in 1,668 individuals with early HD followed up prospectively for up to 30 to 36 months of longitudinal clinical follow-up.
RESULTS:The results demonstrated that a composite measure of motor, cognitive, and global functional decline best characterized clinical progression and was most strongly associated with brain measures of progressive corticostriatal atrophy.
CONCLUSIONS:Use of a composite motor, cognitive, and global functional clinical outcome measure in HD provides an improved measure of clinical progression more related to measures of progressive brain atrophy and provides an opportunity for enhanced clinical trial efficiency relative to currently used individual motor, cognitive, and functional outcome measures.</abstract><cop>United States</cop><pub>American Academy of Neurology</pub><pmid>29142089</pmid><doi>10.1212/WNL.0000000000004743</doi><tpages>8</tpages><oa>free_for_read</oa></addata></record> |
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| ispartof | Neurology, 2017-12, Vol.89 (24), p.2495-2502 |
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| source | MEDLINE; Alma/SFX Local Collection; Journals@Ovid Complete |
| subjects | Adult Apathy Cognitive Dysfunction - physiopathology Cognitive Dysfunction - psychology Disease Progression Emotions Facial Recognition Female Humans Huntington Disease - drug therapy Huntington Disease - physiopathology Huntington Disease - psychology Longitudinal Studies Male Middle Aged Motor Skills Neuropsychological Tests Prospective Studies Randomized Controlled Trials as Topic Reproducibility of Results Signal-To-Noise Ratio Social Perception Stroop Test Ubiquinone - analogs & derivatives Ubiquinone - therapeutic use Vitamins - therapeutic use |
| title | Motor, cognitive, and functional declines contribute to a single progressive factor in early HD |
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