Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.

<h4>Introduction</h4>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...

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Autores principales:	Sean C Forbes, Rebecca J Willcocks, William T Triplett, William D Rooney, Donovan J Lott, Dah-Jyuu Wang, Jim Pollaro, Claudia R Senesac, Michael J Daniels, Richard S Finkel, Barry S Russman, Barry J Byrne, Erika L Finanger, Gihan I Tennekoon, Glenn A Walter, H Lee Sweeney, Krista Vandenborne
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2014
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/b1ddd295609e4a84bb30590f29b6277c
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spelling	oai:doaj.org-article:b1ddd295609e4a84bb30590f29b6277c2021-11-25T06:01:18ZMagnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.1932-620310.1371/journal.pone.0106435https://doaj.org/article/b1ddd295609e4a84bb30590f29b6277c2014-01-01T00:00:00Zhttps://doi.org/10.1371/journal.pone.0106435https://doaj.org/toc/1932-6203<h4>Introduction</h4>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.<h4>Methods</h4>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.<h4>Results</h4>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.<h4>Discussion</h4>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.Sean C ForbesRebecca J WillcocksWilliam T TriplettWilliam D RooneyDonovan J LottDah-Jyuu WangJim PollaroClaudia R SenesacMichael J DanielsRichard S FinkelBarry S RussmanBarry J ByrneErika L FinangerGihan I TennekoonGlenn A WalterH Lee SweeneyKrista VandenbornePublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 9, Iss 9, p e106435 (2014)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Sean C Forbes Rebecca J Willcocks William T Triplett William D Rooney Donovan J Lott Dah-Jyuu Wang Jim Pollaro Claudia R Senesac Michael J Daniels Richard S Finkel Barry S Russman Barry J Byrne Erika L Finanger Gihan I Tennekoon Glenn A Walter H Lee Sweeney Krista Vandenborne Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.
description	<h4>Introduction</h4>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.<h4>Methods</h4>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.<h4>Results</h4>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.<h4>Discussion</h4>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.
format	article
author	Sean C Forbes Rebecca J Willcocks William T Triplett William D Rooney Donovan J Lott Dah-Jyuu Wang Jim Pollaro Claudia R Senesac Michael J Daniels Richard S Finkel Barry S Russman Barry J Byrne Erika L Finanger Gihan I Tennekoon Glenn A Walter H Lee Sweeney Krista Vandenborne
author_facet	Sean C Forbes Rebecca J Willcocks William T Triplett William D Rooney Donovan J Lott Dah-Jyuu Wang Jim Pollaro Claudia R Senesac Michael J Daniels Richard S Finkel Barry S Russman Barry J Byrne Erika L Finanger Gihan I Tennekoon Glenn A Walter H Lee Sweeney Krista Vandenborne
author_sort	Sean C Forbes
title	Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.
title_short	Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.
title_full	Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.
title_fullStr	Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.
title_full_unstemmed	Magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with Duchenne muscular dystrophy: a multicenter cross sectional study.
title_sort	magnetic resonance imaging and spectroscopy assessment of lower extremity skeletal muscles in boys with duchenne muscular dystrophy: a multicenter cross sectional study.
publisher	Public Library of Science (PLoS)
publishDate	2014
url	https://doaj.org/article/b1ddd295609e4a84bb30590f29b6277c
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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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