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Cmah-dystrophin deficient mdx mice display an accelerated cardiac phenotype that is improved following peptide-PMO exon skipping treatment

Abstract:

Duchenne muscular dystrophy (DMD) is caused by loss of dystrophin protein, leading to progressive muscle weakness and premature death due to respiratory and/or cardiac complications. Cardiac involvement is characterized by progressive dilated cardiomyopathy, decreased fractional shortening and metabolic dysfunction involving reduced metabolism of fatty acids-the major cardiac metabolic substrate. Several mouse models have been developed to study molecular and pathological consequences of dyst...

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Publication status:
Published
Peer review status:
Peer reviewed
Version:
Publisher's Version

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Publisher copy:
10.1093/hmg/ddy346

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Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Physiology Anatomy and Genetics
Oxford college:
Somerville College
Role:
Author
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Physiology Anatomy and Genetics
Role:
Author
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Physiology Anatomy and Genetics
Role:
Author
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Paediatrics
Role:
Author
ORCID:
0000-0002-5640-7569
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Physiology Anatomy and Genetics
Role:
Author
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Publisher:
Oxford University Press Publisher's website
Journal:
Human Molecular Genetics Journal website
Volume:
28
Issue:
3
Pages:
396–406
Publication date:
2018-10-02
Acceptance date:
2018-09-21
DOI:
EISSN:
1460-2083
ISSN:
0964-6906
Pubs id:
pubs:923772
URN:
uri:c36deec4-3259-4a35-adf8-7622f9d6708b
UUID:
uuid:c36deec4-3259-4a35-adf8-7622f9d6708b
Local pid:
pubs:923772
Language:
English

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