Functional changes in Duchenne muscular dystrophy: A 12-month longitudinal cohort study
The aim of the study was to assess different outcome measures in a cohort of ambulant boys with Duchenne muscular dystrophy (DMD) over 12 months in order to establish the spectrum of possible changes in relation to age and steroid treatment. The study is a longitudinal multicentric cohort study. A t...
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| Veröffentlicht in: | Neurology 2011-07, Vol.77 (3), p.250-256 |
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| creator | MAZZONE, E VASCO, G FROSINI, S BELLO, L BONFIGLIO, S ZUCCHINI, E DE SANCTIS, R SCUTIFERO, M BIANCO, F ROSSI, F MOTTA, M. C SACCO, A SORMANI, M. P DONATI, M. A MONGINI, T PINI, A BATTINI, R PEGORARO, E PANE, M GASPERINI, S PREVITALI, S NAPOLITANO, S MARTINELLI, D TORRENTE, Y BRUNO, C VITA, G COMI, G BERTINI, E MERCURI, E BERARDINELLI, A MESSINA, S D'AMICO, A DOGLIO, L POLITANO, L CAVALLARO, F |
| description | The aim of the study was to assess different outcome measures in a cohort of ambulant boys with Duchenne muscular dystrophy (DMD) over 12 months in order to establish the spectrum of possible changes in relation to age and steroid treatment.
The study is a longitudinal multicentric cohort study. A total of 106 ambulant patients with DMD were assessed using the 6-minute walk test (6MWT) and North Star Ambulatory Assessment (NSAA) at baseline and 12 months. Clinical data including age and steroid treatment were collected.
During the 12 months of the study, we observed a mean decline of 25.8 meters in the 6MWT with a SD of 74.3 meters. On NSAA, the mean decline was 2.2 points with a SD of 3.7. Not all the boys with DMD in our cohort showed a decline over the 12 months, with young boys showing some improvement in their 6MWT and NSAA scores up to the age of 7. NSAA and the 6MWT had the highest correlation (r = 0.52, p < 0.001).
This study provides longitudinal data of NSAA and 6MWT over a 12-month period. These data can be useful when designing a clinical trial. |
| doi_str_mv | 10.1212/WNL.0b013e318225ab2e |
| format | Article |
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The study is a longitudinal multicentric cohort study. A total of 106 ambulant patients with DMD were assessed using the 6-minute walk test (6MWT) and North Star Ambulatory Assessment (NSAA) at baseline and 12 months. Clinical data including age and steroid treatment were collected.
During the 12 months of the study, we observed a mean decline of 25.8 meters in the 6MWT with a SD of 74.3 meters. On NSAA, the mean decline was 2.2 points with a SD of 3.7. Not all the boys with DMD in our cohort showed a decline over the 12 months, with young boys showing some improvement in their 6MWT and NSAA scores up to the age of 7. NSAA and the 6MWT had the highest correlation (r = 0.52, p < 0.001).
This study provides longitudinal data of NSAA and 6MWT over a 12-month period. These data can be useful when designing a clinical trial.</description><identifier>ISSN: 0028-3878</identifier><identifier>EISSN: 1526-632X</identifier><identifier>DOI: 10.1212/WNL.0b013e318225ab2e</identifier><identifier>PMID: 21734183</identifier><identifier>CODEN: NEURAI</identifier><language>eng</language><publisher>Hagerstown, MD: Lippincott Williams & Wilkins</publisher><subject>Adolescent ; Anti-Inflammatory Agents - therapeutic use ; Biological and medical sciences ; Child ; Child, Preschool ; Cohort Studies ; Cross-Sectional Studies ; Disease Progression ; Diseases of striated muscles. Neuromuscular diseases ; Female ; Humans ; Male ; Medical sciences ; Muscular Dystrophy, Duchenne - drug therapy ; Muscular Dystrophy, Duchenne - physiopathology ; Neurology ; Prednisolone - therapeutic use ; Pregnenediones - therapeutic use ; Reproducibility of Results ; Severity of Illness Index ; Statistics as Topic ; Walking - physiology</subject><ispartof>Neurology, 2011-07, Vol.77 (3), p.250-256</ispartof><rights>2015 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c241t-b44f2e56181d3ca9c595cff9ea5e85016d6bf596ce70c590ae9627280675d2483</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,27901,27902</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=24365820$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/21734183$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>MAZZONE, E</creatorcontrib><creatorcontrib>VASCO, G</creatorcontrib><creatorcontrib>FROSINI, S</creatorcontrib><creatorcontrib>BELLO, L</creatorcontrib><creatorcontrib>BONFIGLIO, S</creatorcontrib><creatorcontrib>ZUCCHINI, E</creatorcontrib><creatorcontrib>DE SANCTIS, R</creatorcontrib><creatorcontrib>SCUTIFERO, M</creatorcontrib><creatorcontrib>BIANCO, F</creatorcontrib><creatorcontrib>ROSSI, F</creatorcontrib><creatorcontrib>MOTTA, M. C</creatorcontrib><creatorcontrib>SACCO, A</creatorcontrib><creatorcontrib>SORMANI, M. P</creatorcontrib><creatorcontrib>DONATI, M. A</creatorcontrib><creatorcontrib>MONGINI, T</creatorcontrib><creatorcontrib>PINI, A</creatorcontrib><creatorcontrib>BATTINI, R</creatorcontrib><creatorcontrib>PEGORARO, E</creatorcontrib><creatorcontrib>PANE, M</creatorcontrib><creatorcontrib>GASPERINI, S</creatorcontrib><creatorcontrib>PREVITALI, S</creatorcontrib><creatorcontrib>NAPOLITANO, S</creatorcontrib><creatorcontrib>MARTINELLI, D</creatorcontrib><creatorcontrib>TORRENTE, Y</creatorcontrib><creatorcontrib>BRUNO, C</creatorcontrib><creatorcontrib>VITA, G</creatorcontrib><creatorcontrib>COMI, G</creatorcontrib><creatorcontrib>BERTINI, E</creatorcontrib><creatorcontrib>MERCURI, E</creatorcontrib><creatorcontrib>BERARDINELLI, A</creatorcontrib><creatorcontrib>MESSINA, S</creatorcontrib><creatorcontrib>D'AMICO, A</creatorcontrib><creatorcontrib>DOGLIO, L</creatorcontrib><creatorcontrib>POLITANO, L</creatorcontrib><creatorcontrib>CAVALLARO, F</creatorcontrib><title>Functional changes in Duchenne muscular dystrophy: A 12-month longitudinal cohort study</title><title>Neurology</title><addtitle>Neurology</addtitle><description>The aim of the study was to assess different outcome measures in a cohort of ambulant boys with Duchenne muscular dystrophy (DMD) over 12 months in order to establish the spectrum of possible changes in relation to age and steroid treatment.
The study is a longitudinal multicentric cohort study. A total of 106 ambulant patients with DMD were assessed using the 6-minute walk test (6MWT) and North Star Ambulatory Assessment (NSAA) at baseline and 12 months. Clinical data including age and steroid treatment were collected.
During the 12 months of the study, we observed a mean decline of 25.8 meters in the 6MWT with a SD of 74.3 meters. On NSAA, the mean decline was 2.2 points with a SD of 3.7. Not all the boys with DMD in our cohort showed a decline over the 12 months, with young boys showing some improvement in their 6MWT and NSAA scores up to the age of 7. NSAA and the 6MWT had the highest correlation (r = 0.52, p < 0.001).
This study provides longitudinal data of NSAA and 6MWT over a 12-month period. These data can be useful when designing a clinical trial.</description><subject>Adolescent</subject><subject>Anti-Inflammatory Agents - therapeutic use</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cohort Studies</subject><subject>Cross-Sectional Studies</subject><subject>Disease Progression</subject><subject>Diseases of striated muscles. Neuromuscular diseases</subject><subject>Female</subject><subject>Humans</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Muscular Dystrophy, Duchenne - drug therapy</subject><subject>Muscular Dystrophy, Duchenne - physiopathology</subject><subject>Neurology</subject><subject>Prednisolone - therapeutic use</subject><subject>Pregnenediones - therapeutic use</subject><subject>Reproducibility of Results</subject><subject>Severity of Illness Index</subject><subject>Statistics as Topic</subject><subject>Walking - physiology</subject><issn>0028-3878</issn><issn>1526-632X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2011</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpdkD1PwzAURS0EoqXwDxDywphiP3_EYasKBaQKFlDZIsdxmqDEqexkyL8n0AIS09PVvecNB6FLSuYUKNxsntdzkhHKLKMKQOgM7BGaUgEykgzej9GUEFARU7GaoLMQPggZyzg5RROgMeNUsSnarHpnuqp1usam1G5rA64cvutNaZ2zuOmD6WvtcT6Ezre7crjFC0whalrXlbhu3bbq-rz65tuy9R0OYx7O0Umh62AvDneG3lb3r8vHaP3y8LRcrCMDnHZRxnkBVkiqaM6MToxIhCmKxGphlSBU5jIrRCKNjcnYEW0TCTEoImORA1dshvj-r_FtCN4W6c5XjfZDSkn65SkdPaX_PY3Y1R7b9Vlj81_oR8w4uD4MdDC6Lrx2pgp_O86kUEDYJxlccoc</recordid><startdate>20110719</startdate><enddate>20110719</enddate><creator>MAZZONE, E</creator><creator>VASCO, G</creator><creator>FROSINI, S</creator><creator>BELLO, L</creator><creator>BONFIGLIO, S</creator><creator>ZUCCHINI, E</creator><creator>DE SANCTIS, R</creator><creator>SCUTIFERO, M</creator><creator>BIANCO, F</creator><creator>ROSSI, F</creator><creator>MOTTA, M. C</creator><creator>SACCO, A</creator><creator>SORMANI, M. P</creator><creator>DONATI, M. 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C ; SACCO, A ; SORMANI, M. P ; DONATI, M. A ; MONGINI, T ; PINI, A ; BATTINI, R ; PEGORARO, E ; PANE, M ; GASPERINI, S ; PREVITALI, S ; NAPOLITANO, S ; MARTINELLI, D ; TORRENTE, Y ; BRUNO, C ; VITA, G ; COMI, G ; BERTINI, E ; MERCURI, E ; BERARDINELLI, A ; MESSINA, S ; D'AMICO, A ; DOGLIO, L ; POLITANO, L ; CAVALLARO, F</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c241t-b44f2e56181d3ca9c595cff9ea5e85016d6bf596ce70c590ae9627280675d2483</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2011</creationdate><topic>Adolescent</topic><topic>Anti-Inflammatory Agents - therapeutic use</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Cohort Studies</topic><topic>Cross-Sectional Studies</topic><topic>Disease Progression</topic><topic>Diseases of striated muscles. Neuromuscular diseases</topic><topic>Female</topic><topic>Humans</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Muscular Dystrophy, Duchenne - drug therapy</topic><topic>Muscular Dystrophy, Duchenne - physiopathology</topic><topic>Neurology</topic><topic>Prednisolone - therapeutic use</topic><topic>Pregnenediones - therapeutic use</topic><topic>Reproducibility of Results</topic><topic>Severity of Illness Index</topic><topic>Statistics as Topic</topic><topic>Walking - physiology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>MAZZONE, E</creatorcontrib><creatorcontrib>VASCO, G</creatorcontrib><creatorcontrib>FROSINI, S</creatorcontrib><creatorcontrib>BELLO, L</creatorcontrib><creatorcontrib>BONFIGLIO, S</creatorcontrib><creatorcontrib>ZUCCHINI, E</creatorcontrib><creatorcontrib>DE SANCTIS, R</creatorcontrib><creatorcontrib>SCUTIFERO, M</creatorcontrib><creatorcontrib>BIANCO, F</creatorcontrib><creatorcontrib>ROSSI, F</creatorcontrib><creatorcontrib>MOTTA, M. 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C</au><au>SACCO, A</au><au>SORMANI, M. P</au><au>DONATI, M. A</au><au>MONGINI, T</au><au>PINI, A</au><au>BATTINI, R</au><au>PEGORARO, E</au><au>PANE, M</au><au>GASPERINI, S</au><au>PREVITALI, S</au><au>NAPOLITANO, S</au><au>MARTINELLI, D</au><au>TORRENTE, Y</au><au>BRUNO, C</au><au>VITA, G</au><au>COMI, G</au><au>BERTINI, E</au><au>MERCURI, E</au><au>BERARDINELLI, A</au><au>MESSINA, S</au><au>D'AMICO, A</au><au>DOGLIO, L</au><au>POLITANO, L</au><au>CAVALLARO, F</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Functional changes in Duchenne muscular dystrophy: A 12-month longitudinal cohort study</atitle><jtitle>Neurology</jtitle><addtitle>Neurology</addtitle><date>2011-07-19</date><risdate>2011</risdate><volume>77</volume><issue>3</issue><spage>250</spage><epage>256</epage><pages>250-256</pages><issn>0028-3878</issn><eissn>1526-632X</eissn><coden>NEURAI</coden><abstract>The aim of the study was to assess different outcome measures in a cohort of ambulant boys with Duchenne muscular dystrophy (DMD) over 12 months in order to establish the spectrum of possible changes in relation to age and steroid treatment.
The study is a longitudinal multicentric cohort study. A total of 106 ambulant patients with DMD were assessed using the 6-minute walk test (6MWT) and North Star Ambulatory Assessment (NSAA) at baseline and 12 months. Clinical data including age and steroid treatment were collected.
During the 12 months of the study, we observed a mean decline of 25.8 meters in the 6MWT with a SD of 74.3 meters. On NSAA, the mean decline was 2.2 points with a SD of 3.7. Not all the boys with DMD in our cohort showed a decline over the 12 months, with young boys showing some improvement in their 6MWT and NSAA scores up to the age of 7. NSAA and the 6MWT had the highest correlation (r = 0.52, p < 0.001).
This study provides longitudinal data of NSAA and 6MWT over a 12-month period. These data can be useful when designing a clinical trial.</abstract><cop>Hagerstown, MD</cop><pub>Lippincott Williams & Wilkins</pub><pmid>21734183</pmid><doi>10.1212/WNL.0b013e318225ab2e</doi><tpages>7</tpages></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 0028-3878 |
| ispartof | Neurology, 2011-07, Vol.77 (3), p.250-256 |
| issn | 0028-3878 1526-632X |
| language | eng |
| recordid | cdi_crossref_primary_10_1212_WNL_0b013e318225ab2e |
| source | MEDLINE; Alma/SFX Local Collection; Journals@Ovid Complete |
| subjects | Adolescent Anti-Inflammatory Agents - therapeutic use Biological and medical sciences Child Child, Preschool Cohort Studies Cross-Sectional Studies Disease Progression Diseases of striated muscles. Neuromuscular diseases Female Humans Male Medical sciences Muscular Dystrophy, Duchenne - drug therapy Muscular Dystrophy, Duchenne - physiopathology Neurology Prednisolone - therapeutic use Pregnenediones - therapeutic use Reproducibility of Results Severity of Illness Index Statistics as Topic Walking - physiology |
| title | Functional changes in Duchenne muscular dystrophy: A 12-month longitudinal cohort study |
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