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The cooperative int...
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Henricson, E. K.
(author)
The cooperative international neuromuscular research group Duchenne natural history study: Glucocorticoid treatment preserves clinically meaningful functional milestones and reduces rate of disease progression as measured by manual muscle testing and other commonly used clinical trial outcome measures
- Article/chapterEnglish2013
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LIBRIS-ID:oai:gup.ub.gu.se/181054
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https://gup.ub.gu.se/publication/181054URI
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https://doi.org/10.1002/mus.23808DOI
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Subject category:ref swepub-contenttype
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Subject category:art swepub-publicationtype
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Introduction: Glucocorticoid (GC) therapy in Duchenne muscular dystrophy (DMD) has altered disease progression, necessitating contemporary natural history studies. Methods: The Cooperative Neuromuscular Research Group (CINRG) DMD Natural History Study (DMD-NHS) enrolled 340 DMD males, ages 2-28 years. A comprehensive battery of measures was obtained. Results: A novel composite functional milestone scale scale showed clinically meaningful mobility and upper limb abilities were significantly preserved in GC-treated adolescents/young adults. Manual muscle test (MMT)-based calculations of global strength showed that those patients <10 years of age treated with steroids declined by 0.4 +/- 0.39 MMT unit/year, compared with -0.4 +/- 0.39 MMT unit/year in historical steroid-naive subjects. Pulmonary function tests (PFTs) were relatively preserved in steroid-treated adolescents. The linearity and magnitude of decline in measures were affected by maturational changes and functional status. Conclusions: In DMD, long-term use of GCs showed reduced strength loss and preserved functional capabilities and PFTs compared with previous natural history studies performed prior to the widespread use of GC therapy.
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Abresch, R. T.
(author)
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Cnaan, A.
(author)
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Hu, F. M.
(author)
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Duong, T.
(author)
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Arrieta, A.
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Han, J.
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Escolar, D. M.
(author)
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Florence, J. M.
(author)
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Clemens, P. R.
(author)
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Hoffman, E. P.
(author)
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McDonald, C. M.
(author)
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Tulinius, Mar,1953Gothenburg University,Göteborgs universitet,Institutionen för kliniska vetenskaper, Avdelningen för pediatrik,Institute of Clinical Sciences, Department of Pediatrics(Swepub:gu)xtulim
(author)
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Göteborgs universitetInstitutionen för kliniska vetenskaper, Avdelningen för pediatrik
(creator_code:org_t)
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In:Muscle & Nerve: Wiley48:1, s. 55-670148-639X
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Henricson, E. K.
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Abresch, R. T.
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Cnaan, A.
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Hu, F. M.
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Duong, T.
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Arrieta, A.
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Han, J.
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Escolar, D. M.
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Florence, J. M.
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Clemens, P. R.
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Hoffman, E. P.
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McDonald, C. M.
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Tulinius, Mar, 1 ...
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Muscle & Nerve
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University of Gothenburg