Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study
Duchenne muscular dystrophy (DMD) is an X-linked recessive disorder that results in functional deficits. However, these functional declines are often not able to be quantified in clinical trials for DMD until after age 7. In this study, we hypothesized that (1)H2O T2 derived using (1)H-MRS and MRI-T...
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| creator | Forbes, Sean C Willcocks, Rebecca J Triplett, William T Rooney, William D Lott, Donovan J Wang, Dah-Jyuu Pollaro, Jim Senesac, Claudia R Daniels, Michael J Finkel, Richard S Russman, Barry S Byrne, Barry J Finanger, Erika L Tennekoon, Gihan I Walter, Glenn A Sweeney, H Lee Vandenborne, Krista |
| description | Duchenne muscular dystrophy (DMD) is an X-linked recessive disorder that results in functional deficits. However, these functional declines are often not able to be quantified in clinical trials for DMD until after age 7. In this study, we hypothesized that (1)H2O T2 derived using (1)H-MRS and MRI-T2 will be sensitive to muscle involvement at a young age (5-7 years) consistent with increased inflammation and muscle damage in a large cohort of DMD subjects compared to controls.
MR data were acquired from 123 boys with DMD (ages 5-14 years; mean 8.6 SD 2.2 years) and 31 healthy controls (age 9.7 SD 2.3 years) using 3-Tesla MRI instruments at three institutions (University of Florida, Oregon Health & Science University, and Children's Hospital of Philadelphia). T2-weighted multi-slice spin echo (SE) axial images and single voxel 1H-MRS were acquired from the lower leg and thigh to measure lipid fraction and (1)H2O T2.
MRI-T2, (1)H2O T2, and lipid fraction were greater (p |
| doi_str_mv | 10.1371/journal.pone.0106435 |
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MR data were acquired from 123 boys with DMD (ages 5-14 years; mean 8.6 SD 2.2 years) and 31 healthy controls (age 9.7 SD 2.3 years) using 3-Tesla MRI instruments at three institutions (University of Florida, Oregon Health & Science University, and Children's Hospital of Philadelphia). T2-weighted multi-slice spin echo (SE) axial images and single voxel 1H-MRS were acquired from the lower leg and thigh to measure lipid fraction and (1)H2O T2.
MRI-T2, (1)H2O T2, and lipid fraction were greater (p<0.05) in DMD compared to controls. In the youngest age group, DMD values were different (p<0.05) than controls for the soleus MRI-T2, (1)H2O T2 and lipid fraction and vastus lateralis MRI-T2 and (1)H2O T2. In the boys with DMD, MRI-T2 and lipid fraction were greater (p<0.05) in the oldest age group (11-14 years) than the youngest age group (5-6.9 years), while 1H2O T2 was lower in the oldest age group compared to the young age group.
Overall, MR measures of T2 and lipid fraction revealed differences between DMD and Controls. Furthermore, MRI-T2 was greater in the older age group compared to the young age group, which was associated with higher lipid fractions. Overall, MR measures of T2 and lipid fraction show excellent sensitivity to DMD disease pathologies and potential therapeutic interventions in DMD, even in the younger boys.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0106435</identifier><identifier>PMID: 25203313</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Adolescent ; Age ; Age Distribution ; Case-Control Studies ; Child ; Child, Preschool ; Children ; Children & youth ; Clinical trials ; Cohort Studies ; Control equipment ; Control methods ; Cross-Sectional Studies ; Data acquisition ; Disease Progression ; Duchenne's muscular dystrophy ; Dystrophy ; Edema ; Hereditary diseases ; Hospitals ; Humans ; Image acquisition ; Leg ; Lipid Metabolism ; Lipids ; Magnetic resonance ; Magnetic Resonance Imaging ; Male ; Medical research ; Medicine and Health Sciences ; Muscle, Skeletal - metabolism ; Muscles ; Muscular dystrophy ; Muscular Dystrophy, Duchenne - metabolism ; NMR ; Nuclear magnetic resonance ; Physical therapy ; Physiology ; Principal components analysis ; Rodents ; Science ; Skeletal muscle ; Spectroscopy ; Spectrum analysis ; Therapeutic applications ; Thigh</subject><ispartof>PloS one, 2014-09, Vol.9 (9), p.e106435-e106435</ispartof><rights>2014 Forbes et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>2014 Forbes et al 2014 Forbes et al</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c526t-6bc26edc594e86198eca50c51537f934f2c0804f9fb70ad649fdcff8e96dc71a3</citedby><cites>FETCH-LOGICAL-c526t-6bc26edc594e86198eca50c51537f934f2c0804f9fb70ad649fdcff8e96dc71a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC4159278/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC4159278/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,860,881,2096,2915,23845,27901,27902,53766,53768,79343,79344</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/25203313$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><contributor>Taneja, Reshma</contributor><creatorcontrib>Forbes, Sean C</creatorcontrib><creatorcontrib>Willcocks, Rebecca J</creatorcontrib><creatorcontrib>Triplett, William T</creatorcontrib><creatorcontrib>Rooney, William D</creatorcontrib><creatorcontrib>Lott, Donovan J</creatorcontrib><creatorcontrib>Wang, Dah-Jyuu</creatorcontrib><creatorcontrib>Pollaro, Jim</creatorcontrib><creatorcontrib>Senesac, Claudia R</creatorcontrib><creatorcontrib>Daniels, Michael J</creatorcontrib><creatorcontrib>Finkel, Richard S</creatorcontrib><creatorcontrib>Russman, Barry S</creatorcontrib><creatorcontrib>Byrne, Barry J</creatorcontrib><creatorcontrib>Finanger, Erika L</creatorcontrib><creatorcontrib>Tennekoon, Gihan I</creatorcontrib><creatorcontrib>Walter, Glenn A</creatorcontrib><creatorcontrib>Sweeney, H Lee</creatorcontrib><creatorcontrib>Vandenborne, Krista</creatorcontrib><title>Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study</title><title>PloS one</title><addtitle>PLoS One</addtitle><description>Duchenne muscular dystrophy (DMD) is an X-linked recessive disorder that results in functional deficits. However, these functional declines are often not able to be quantified in clinical trials for DMD until after age 7. In this study, we hypothesized that (1)H2O T2 derived using (1)H-MRS and MRI-T2 will be sensitive to muscle involvement at a young age (5-7 years) consistent with increased inflammation and muscle damage in a large cohort of DMD subjects compared to controls.
MR data were acquired from 123 boys with DMD (ages 5-14 years; mean 8.6 SD 2.2 years) and 31 healthy controls (age 9.7 SD 2.3 years) using 3-Tesla MRI instruments at three institutions (University of Florida, Oregon Health & Science University, and Children's Hospital of Philadelphia). T2-weighted multi-slice spin echo (SE) axial images and single voxel 1H-MRS were acquired from the lower leg and thigh to measure lipid fraction and (1)H2O T2.
MRI-T2, (1)H2O T2, and lipid fraction were greater (p<0.05) in DMD compared to controls. In the youngest age group, DMD values were different (p<0.05) than controls for the soleus MRI-T2, (1)H2O T2 and lipid fraction and vastus lateralis MRI-T2 and (1)H2O T2. In the boys with DMD, MRI-T2 and lipid fraction were greater (p<0.05) in the oldest age group (11-14 years) than the youngest age group (5-6.9 years), while 1H2O T2 was lower in the oldest age group compared to the young age group.
Overall, MR measures of T2 and lipid fraction revealed differences between DMD and Controls. Furthermore, MRI-T2 was greater in the older age group compared to the young age group, which was associated with higher lipid fractions. Overall, MR measures of T2 and lipid fraction show excellent sensitivity to DMD disease pathologies and potential therapeutic interventions in DMD, even in the younger boys.</description><subject>Adolescent</subject><subject>Age</subject><subject>Age Distribution</subject><subject>Case-Control Studies</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Children</subject><subject>Children & youth</subject><subject>Clinical trials</subject><subject>Cohort Studies</subject><subject>Control equipment</subject><subject>Control methods</subject><subject>Cross-Sectional Studies</subject><subject>Data acquisition</subject><subject>Disease Progression</subject><subject>Duchenne's muscular dystrophy</subject><subject>Dystrophy</subject><subject>Edema</subject><subject>Hereditary diseases</subject><subject>Hospitals</subject><subject>Humans</subject><subject>Image acquisition</subject><subject>Leg</subject><subject>Lipid Metabolism</subject><subject>Lipids</subject><subject>Magnetic resonance</subject><subject>Magnetic Resonance Imaging</subject><subject>Male</subject><subject>Medical research</subject><subject>Medicine and Health Sciences</subject><subject>Muscle, Skeletal - metabolism</subject><subject>Muscles</subject><subject>Muscular dystrophy</subject><subject>Muscular Dystrophy, Duchenne - metabolism</subject><subject>NMR</subject><subject>Nuclear magnetic resonance</subject><subject>Physical therapy</subject><subject>Physiology</subject><subject>Principal components analysis</subject><subject>Rodents</subject><subject>Science</subject><subject>Skeletal muscle</subject><subject>Spectroscopy</subject><subject>Spectrum analysis</subject><subject>Therapeutic applications</subject><subject>Thigh</subject><issn>1932-6203</issn><issn>1932-6203</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2014</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><sourceid>DOA</sourceid><recordid>eNptUstu1DAUjRCIloE_QGCJDZsZ7PiRmEUlVF6VitjA2nKc65kMjh3shJI_4jNxZ9KqRaxsXZ977rnHpyieE7whtCJv9mGKXrvNEDxsMMGCUf6gOCWSlmtRYvrwzv2keJLSHmNOayEeFyclz0VK6Gnx54veehg7gyKk4LU3gLpebzu_Rdq3KA1gxhiSCcOMdEqQUg9-RMEiF64gIvg9Rui7cUbpBzgYtUP9lIyDhDqPmjAndNWNO_R-MjvwHg6vk9MRtXPKzMNufot0rrosIjNnSpPnJZTy4C4rciiNUzs_LR5Z7RI8W85V8f3jh2_nn9eXXz9dnL-7XBteinEtGlMKaA2XDGpBZA1Gc2w44bSykjJbGlxjZqVtKqxbwaRtjbU1SNGaimi6Kl4eeQcXklpMTopwQTCjJPu2Ki6OiDbovRpitivOKuhOHQohbpWOeRkHqiFt25aSCyyB6Zo1DcVcYlvKRpRVZTLX2TJtavosOxsQtbtHev_Fdzu1Db8UI1yWVZ0JXi8EMfycII2q75IB57SHMB11M8Hrmmfoq3-g_9-OHVGHX4hgb8UQrK6Dd9OlroOnluDlthd3F7ltukka_QuLF9yS</recordid><startdate>20140909</startdate><enddate>20140909</enddate><creator>Forbes, Sean 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resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study</title><author>Forbes, Sean C ; Willcocks, Rebecca J ; Triplett, William T ; Rooney, William D ; Lott, Donovan J ; Wang, Dah-Jyuu ; Pollaro, Jim ; Senesac, Claudia R ; Daniels, Michael J ; Finkel, Richard S ; Russman, Barry S ; Byrne, Barry J ; Finanger, Erika L ; Tennekoon, Gihan I ; Walter, Glenn A ; Sweeney, H Lee ; Vandenborne, Krista</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c526t-6bc26edc594e86198eca50c51537f934f2c0804f9fb70ad649fdcff8e96dc71a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2014</creationdate><topic>Adolescent</topic><topic>Age</topic><topic>Age Distribution</topic><topic>Case-Control Studies</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Children</topic><topic>Children & 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Jim</au><au>Senesac, Claudia R</au><au>Daniels, Michael J</au><au>Finkel, Richard S</au><au>Russman, Barry S</au><au>Byrne, Barry J</au><au>Finanger, Erika L</au><au>Tennekoon, Gihan I</au><au>Walter, Glenn A</au><au>Sweeney, H Lee</au><au>Vandenborne, Krista</au><au>Taneja, Reshma</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study</atitle><jtitle>PloS one</jtitle><addtitle>PLoS One</addtitle><date>2014-09-09</date><risdate>2014</risdate><volume>9</volume><issue>9</issue><spage>e106435</spage><epage>e106435</epage><pages>e106435-e106435</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>Duchenne muscular dystrophy (DMD) is an X-linked recessive disorder that results in functional deficits. However, these functional declines are often not able to be quantified in clinical trials for DMD until after age 7. In this study, we hypothesized that (1)H2O T2 derived using (1)H-MRS and MRI-T2 will be sensitive to muscle involvement at a young age (5-7 years) consistent with increased inflammation and muscle damage in a large cohort of DMD subjects compared to controls.
MR data were acquired from 123 boys with DMD (ages 5-14 years; mean 8.6 SD 2.2 years) and 31 healthy controls (age 9.7 SD 2.3 years) using 3-Tesla MRI instruments at three institutions (University of Florida, Oregon Health & Science University, and Children's Hospital of Philadelphia). T2-weighted multi-slice spin echo (SE) axial images and single voxel 1H-MRS were acquired from the lower leg and thigh to measure lipid fraction and (1)H2O T2.
MRI-T2, (1)H2O T2, and lipid fraction were greater (p<0.05) in DMD compared to controls. In the youngest age group, DMD values were different (p<0.05) than controls for the soleus MRI-T2, (1)H2O T2 and lipid fraction and vastus lateralis MRI-T2 and (1)H2O T2. In the boys with DMD, MRI-T2 and lipid fraction were greater (p<0.05) in the oldest age group (11-14 years) than the youngest age group (5-6.9 years), while 1H2O T2 was lower in the oldest age group compared to the young age group.
Overall, MR measures of T2 and lipid fraction revealed differences between DMD and Controls. Furthermore, MRI-T2 was greater in the older age group compared to the young age group, which was associated with higher lipid fractions. Overall, MR measures of T2 and lipid fraction show excellent sensitivity to DMD disease pathologies and potential therapeutic interventions in DMD, even in the younger boys.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>25203313</pmid><doi>10.1371/journal.pone.0106435</doi><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 1932-6203 |
| ispartof | PloS one, 2014-09, Vol.9 (9), p.e106435-e106435 |
| issn | 1932-6203 1932-6203 |
| language | eng |
| recordid | cdi_plos_journals_1561043120 |
| source | Public Library of Science (PLoS) Journals Open Access; MEDLINE; DOAJ Directory of Open Access Journals; EZB-FREE-00999 freely available EZB journals; PubMed Central; Free Full-Text Journals in Chemistry |
| subjects | Adolescent Age Age Distribution Case-Control Studies Child Child, Preschool Children Children & youth Clinical trials Cohort Studies Control equipment Control methods Cross-Sectional Studies Data acquisition Disease Progression Duchenne's muscular dystrophy Dystrophy Edema Hereditary diseases Hospitals Humans Image acquisition Leg Lipid Metabolism Lipids Magnetic resonance Magnetic Resonance Imaging Male Medical research Medicine and Health Sciences Muscle, Skeletal - metabolism Muscles Muscular dystrophy Muscular Dystrophy, Duchenne - metabolism NMR Nuclear magnetic resonance Physical therapy Physiology Principal components analysis Rodents Science Skeletal muscle Spectroscopy Spectrum analysis Therapeutic applications Thigh |
| title | Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study |
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