ArticleDisease models & mechanisms2014
Predictive markers of clinical outcome in the GRMD dog model of Duchenne muscular dystrophy.
Article in Disease models & mechanisms, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers.
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Who cites it
21 citing papers in PubMed, 37 citations in OpenAlex.
- Six decades of animal accelerometry: trends, applications, and future directions.Movement ecology · 2026Review
- Review
- T cell biology in neuromuscular disorders: a focus on Duchenne Muscular Dystrophy and Amyotrophic Lateral Sclerosis.Frontiers in immunology · 2023Review
- Serum inflammatory cytokines as disease biomarkers in the DE50-MD dog model of Duchenne muscular dystrophy.Disease models & mechanisms · 2022Article
- Gene Editing to Tackle Facioscapulohumeral Muscular Dystrophy.Frontiers in genome editing · 2022Review
- Development of outcome measures according to dystrophic phenotypes in canine X-linked muscular dystrophy in Japan.Experimental animals · 2021Article
- The Immune System in Duchenne Muscular Dystrophy Pathogenesis.Biomedicines · 2021Review
- Integrin-directed antibody-based immunotherapy: focus on VLA-4.Immunotherapy advances · 2021Review
- Myostatin Is a Quantifiable Biomarker for Monitoring Pharmaco-gene Therapy in Duchenne Muscular Dystrophy.Molecular therapy. Methods & clinical development · 2020Article
- X-linked muscular dystrophy in a Labrador Retriever strain: phenotypic and molecular characterisation.Skeletal muscle · 2020Article
- miR-379 links glucocorticoid treatment with mitochondrial response in Duchenne muscular dystrophy.Scientific reports · 2020Article
- Crosstalk Between Innate and T Cell Adaptive Immunity With(in) the Muscle.Frontiers in physiology · 2020Review
- Natural History of Cardiomyopathy in Adult Dogs With Golden Retriever Muscular Dystrophy.Journal of the American Heart Association · 2019Article
- The Dog Model in the Spotlight: Legacy of a Trustful Cooperation.Journal of neuromuscular diseases · 2019Article
- Accelerometric outcomes of motor function related to clinical evaluations and muscle involvement in dystrophic dogs.PloS one · 2018Article
- Long-term effects of systemic gene therapy in a canine model of myotubular myopathy.Muscle & nerve · 2017Article
- The golden retriever model of Duchenne muscular dystrophy.Skeletal muscle · 2017Review
- [Research progress on disease models and gene therapy of Duchenne muscular dystrophy].Zhejiang da xue xue bao. Yi xue ban = Journal of Zhejiang University. Medical sciences · 2016Review
- Early loss of ambulation is not a representative clinical feature in Duchenne muscular dystrophy dogs: remarks on the article of Barthélémy et al.Disease models & mechanisms · 2015Article
- CD49d is a disease progression biomarker and a potential target for immunotherapy in Duchenne muscular dystrophy.Skeletal muscle · 2015Article
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Authors and funding
11 authors at 7 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
In the translational process of developing innovative therapies for DMD (Duchenne muscular dystrophy), the last preclinical validation step is often carried out in the most relevant animal model of this human disease, namely the GRMD (Golden Retriever muscular dystrophy) dog. The disease in GRMD dogs mimics human DMD in many aspects, including the inter-individual heterogeneity. This last point can be seen as a drawback for an animal model but is inherently related to the disease in GRMD dogs closely resembling that of individuals with DMD. In order to improve the management of this inter-individual heterogeneity, we have screened a combination of biomarkers in sixty-one 2-month-old GRMD dogs at the onset of the disease and a posteriori we addressed their predictive value on the severity of the disease. Three non-invasive biomarkers obtained at early stages of the disease were found to be highly predictive for the loss of ambulation before 6 months of age. An elevation in the number of circulating CD4(+)CD49d(hi) T cells and a decreased stride frequency resulting in a reduced spontaneous speed were found to be strongly associated with the severe clinical form of the disease. These factors can be used as predictive tests to screen dogs to separate them into groups with slow or fast disease progression before their inclusion into a therapeutic preclinical trial, and therefore improve the reliability and translational value of the trials carried out on this invaluable large animal model. These same biomarkers have also been described to be predictive for the time to loss of ambulation in boys with DMD, strengthening the relevance of GRMD dogs as preclinical models of this devastating muscle disease.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.