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Multiomic characterization of disease progression in mice lacking dystrophin
by
Signorelli, Mirko
, Aartsma-Rus, Annemieke
, Tsonaka, Roula
, Spitali, Pietro
in
Analysis
/ Animal models
/ Animals
/ Biology and Life Sciences
/ Biomarkers
/ Blood
/ Clinical trials
/ Datasets
/ Development and progression
/ Disease Models, Animal
/ Disease Progression
/ Duchenne muscular dystrophy
/ Duchenne's muscular dystrophy
/ Dystrophin
/ Dystrophin - genetics
/ Dystrophin - metabolism
/ Dystrophy
/ Energy metabolism
/ Experiments
/ Factor analysis
/ Gene expression
/ Genes
/ Genetic aspects
/ Health aspects
/ Lipids
/ Mass spectrometry
/ Mass spectroscopy
/ Medicine and Health Sciences
/ Metabolism
/ Metabolites
/ Metabolomics
/ Mice
/ Mice, Inbred mdx
/ Multiomics
/ Muscle, Skeletal - metabolism
/ Muscles
/ Muscular dystrophy
/ Muscular Dystrophy, Duchenne - pathology
/ Plasma
/ Research and Analysis Methods
/ RNA
/ Skeletal muscle
/ Statistical analysis
/ Steroids
/ Trajectory analysis
/ Trends
2023
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Multiomic characterization of disease progression in mice lacking dystrophin
by
Signorelli, Mirko
, Aartsma-Rus, Annemieke
, Tsonaka, Roula
, Spitali, Pietro
in
Analysis
/ Animal models
/ Animals
/ Biology and Life Sciences
/ Biomarkers
/ Blood
/ Clinical trials
/ Datasets
/ Development and progression
/ Disease Models, Animal
/ Disease Progression
/ Duchenne muscular dystrophy
/ Duchenne's muscular dystrophy
/ Dystrophin
/ Dystrophin - genetics
/ Dystrophin - metabolism
/ Dystrophy
/ Energy metabolism
/ Experiments
/ Factor analysis
/ Gene expression
/ Genes
/ Genetic aspects
/ Health aspects
/ Lipids
/ Mass spectrometry
/ Mass spectroscopy
/ Medicine and Health Sciences
/ Metabolism
/ Metabolites
/ Metabolomics
/ Mice
/ Mice, Inbred mdx
/ Multiomics
/ Muscle, Skeletal - metabolism
/ Muscles
/ Muscular dystrophy
/ Muscular Dystrophy, Duchenne - pathology
/ Plasma
/ Research and Analysis Methods
/ RNA
/ Skeletal muscle
/ Statistical analysis
/ Steroids
/ Trajectory analysis
/ Trends
2023
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Do you wish to request the book?
Multiomic characterization of disease progression in mice lacking dystrophin
by
Signorelli, Mirko
, Aartsma-Rus, Annemieke
, Tsonaka, Roula
, Spitali, Pietro
in
Analysis
/ Animal models
/ Animals
/ Biology and Life Sciences
/ Biomarkers
/ Blood
/ Clinical trials
/ Datasets
/ Development and progression
/ Disease Models, Animal
/ Disease Progression
/ Duchenne muscular dystrophy
/ Duchenne's muscular dystrophy
/ Dystrophin
/ Dystrophin - genetics
/ Dystrophin - metabolism
/ Dystrophy
/ Energy metabolism
/ Experiments
/ Factor analysis
/ Gene expression
/ Genes
/ Genetic aspects
/ Health aspects
/ Lipids
/ Mass spectrometry
/ Mass spectroscopy
/ Medicine and Health Sciences
/ Metabolism
/ Metabolites
/ Metabolomics
/ Mice
/ Mice, Inbred mdx
/ Multiomics
/ Muscle, Skeletal - metabolism
/ Muscles
/ Muscular dystrophy
/ Muscular Dystrophy, Duchenne - pathology
/ Plasma
/ Research and Analysis Methods
/ RNA
/ Skeletal muscle
/ Statistical analysis
/ Steroids
/ Trajectory analysis
/ Trends
2023
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Multiomic characterization of disease progression in mice lacking dystrophin
Journal Article
Multiomic characterization of disease progression in mice lacking dystrophin
2023
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Overview
Duchenne muscular dystrophy (DMD) is caused by genetic mutations leading to lack of dystrophin in skeletal muscle. A better understanding of how objective biomarkers for DMD vary across subjects and over time is needed to model disease progression and response to therapy more effectively, both in pre-clinical and clinical research. We present an in-depth characterization of disease progression in 3 murine models of DMD by multiomic analysis of longitudinal trajectories between 6 and 30 weeks of age. Integration of RNA-seq, mass spectrometry-based metabolomic and lipidomic data obtained in muscle and blood samples by Multi-Omics Factor Analysis (MOFA) led to the identification of 8 latent factors that explained 78.8% of the variance in the multiomic dataset. Latent factors could discriminate dystrophic and healthy mice, as well as different time-points. MOFA enabled to connect the gene expression signature in dystrophic muscles, characterized by pro-fibrotic and energy metabolism alterations, to inflammation and lipid signatures in blood. Our results show that omic observations in blood can be directly related to skeletal muscle pathology in dystrophic muscle.
Publisher
Public Library of Science,Public Library of Science (PLoS)
Subject
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