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@ARTICLE{Dowling:259687,
author = {Dowling, Paul and Gargan, Stephen and Zweyer, Margit and
Swandulla, Dieter and Ohlendieck, Kay},
title = {{E}xtracellular {M}atrix {P}roteomics: {T}he mdx-4cv
{M}ouse {D}iaphragm as a {S}urrogate for {S}tudying
{M}yofibrosis in {D}ystrophinopathy.},
journal = {Biomolecules},
volume = {13},
number = {7},
issn = {2218-273X},
address = {Basel},
publisher = {MDPI},
reportid = {DZNE-2023-00759},
pages = {1108},
year = {2023},
abstract = {The progressive degeneration of the skeletal musculature in
Duchenne muscular dystrophy is accompanied by reactive
myofibrosis, fat substitution, and chronic inflammation.
Fibrotic changes and reduced tissue elasticity correlate
with the loss in motor function in this X-chromosomal
disorder. Thus, although dystrophinopathies are due to
primary abnormalities in the DMD gene causing the
almost-complete absence of the cytoskeletal Dp427-M isoform
of dystrophin in voluntary muscles, the excessive
accumulation of extracellular matrix proteins presents a key
histopathological hallmark of muscular dystrophy. Animal
model research has been instrumental in the characterization
of dystrophic muscles and has contributed to a better
understanding of the complex pathogenesis of
dystrophinopathies, the discovery of new disease biomarkers,
and the testing of novel therapeutic strategies. In this
article, we review how mass-spectrometry-based proteomics
can be used to study changes in key components of the
endomysium, perimysium, and epimysium, such as collagens,
proteoglycans, matricellular proteins, and adhesion
receptors. The mdx-4cv mouse diaphragm displays severe
myofibrosis, making it an ideal model system for large-scale
surveys of systematic alterations in the matrisome of
dystrophic fibers. Novel biomarkers of myofibrosis can now
be tested for their appropriateness in the preclinical and
clinical setting as diagnostic, pharmacodynamic, prognostic,
and/or therapeutic monitoring indicators.},
subtyp = {Review Article},
keywords = {Animals / Mice / Mice, Inbred mdx / Diaphragm: metabolism /
Diaphragm: pathology / Proteomics / Muscular Dystrophy,
Duchenne: pathology / Muscle, Skeletal: metabolism /
Extracellular Matrix: metabolism / Extracellular Matrix
Proteins: genetics / Extracellular Matrix Proteins:
metabolism / Biomarkers: metabolism / biglycan (Other) /
collagen (Other) / dystrophin (Other) / dystrophinopathy
(Other) / extracellular matrix (Other) / fibronectin (Other)
/ fibrosis (Other) / matrisome (Other) / mdx (Other) /
periostin (Other) / Extracellular Matrix Proteins (NLM
Chemicals) / Biomarkers (NLM Chemicals)},
cin = {AG Sabir},
ddc = {570},
cid = {I:(DE-2719)5000032},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:37509144},
pmc = {pmc:PMC10377647},
doi = {10.3390/biom13071108},
url = {https://pub.dzne.de/record/259687},
}