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Extraocular muscles (EOMs) are highly specialized skeletal muscles that originate from the head mesoderm and control eye movements. EOMs are uniquely spared in Duchenne muscular dystrophy and animal models of dystrophin deficiency. Specific traits of myogenic progenitors may be determinants of this preferential sparing, but very little is known about the myogenic cells in this muscle group. While satellite cells (SCs) have long been recognized as the main source of myogenic cells in adult muscle, most of the knowledge about these cells comes from the prototypic limb muscles. In this study, we show that EOMs, regardless of their distinctive Pax3-negative lineage origin, harbor SCs that share a common signature (Pax7(+), Ki67(-), Nestin-GFP(+), Myf5(nLacZ+), MyoD-positive lineage origin) with their limb and diaphragm somite-derived counterparts, but are remarkably endowed with a high proliferative potential as revealed in cell culture assays. Specifically, we demonstrate that in adult as well as in aging mice, EOM SCs possess a superior expansion capacity, contributing significantly more proliferating, differentiating and renewal progeny than their limb and diaphragm counterparts. These robust growth and renewal properties are maintained by EOM SCs isolated from dystrophin-null (mdx) mice, while SCs from muscles affected by dystrophin deficiency (i.e., limb and diaphragm) expand poorly in vitro. EOM SCs also retain higher performance in cell transplantation assays in which donor cells were engrafted into host mdx limb muscle. Collectively, our study provides a comprehensive picture of EOM myogenic progenitors, showing that while these cells share common hallmarks with the prototypic SCs in somite-derived muscles, they distinctively feature robust growth and renewal capacities that warrant the title of high performance myo-engines and promote consideration of their properties for developing new approaches in cell-based therapy to combat skeletal muscle wasting.
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http://dx.doi.org/10.1016/j.ydbio.2014.08.035 | DOI Listing |
J Neurochem
September 2025
Department of Biology and Biotechnologies "Charles Darwin", Sapienza University of Rome, Rome, Italy.
Patients with Duchenne muscular dystrophy (DMD) may experience neurobehavioral and cognitive concerns, including psychiatric symptoms, due to the absence of full-length dystrophin (Dp427), frequently accompanied by deficiencies in shorter isoforms. The lack of dystrophin affects neurophysiological processes from the uterine phase, impacting neural circuitry in brain regions such as the prefrontal cortex, hippocampus, and cerebellum. This leads to reduced inhibitory GABAergic transmission and altered hippocampal glutamatergic signaling.
View Article and Find Full Text PDFActa Neurol Belg
September 2025
Department of Pediatric Orthopedics, Chengdu Women's and Children's Central Hospital, School of Medicine, University of Electronic Science and Technology of China, No. 1617, Section 1, Riyue Avenue, Qingyang District, Chengdu, Sichuan Province, China.
Duchenne muscular dystrophy, the most prevalent form of muscular dystrophy, is characterized by neurological complications including cognitive impairment, neuropsychiatric disorders, and epilepsy. Neuroimaging investigations have demonstrated structural brain alterations, hemodynamic disturbances, and metabolic dysregulation in individuals with Duchenne muscular dystrophy. These neurological impairments are primarily attributed to cerebral dystrophin deficiency and subsequent downstream molecular/cellular abnormalities, including altered excitation-inhibition balance, blood-brain barrier disruption, calcium dysregulation, and neuroinflammation.
View Article and Find Full Text PDFMol Ther Nucleic Acids
September 2025
Center for Genetic Medicine Research, Children's National Research Institute, Children's National Research and Innovation Campus, Children's National Hospital, Washington, DC 20012, USA.
Despite the proven safety of dystrophin-targeting phosphorodiamidate morpholino oligomer (PMO) therapy, poor delivery of the PMOs limit the efficacy of this dystrophin restoring gene therapy for Duchenne muscular dystrophy (DMD). Limited myogenesis and excessive fibrosis in DMD are pathological features that contribute to the poor efficacy of PMOs. We show that the severe DMD mouse model (D2-) not only replicates these pathological features of DMD but also mirrors the resulting PMO-mediated dystrophin restoration deficit.
View Article and Find Full Text PDFJ Neuromuscul Dis
August 2025
Department of Human Genetics, Gilbert and Rose-Marie Chagoury School of Medicine, Lebanese American University, Byblos, Lebanon.
Background: Duchenne muscular dystrophy (DMD) is a severe X-linked neuromuscular disorder caused by pathogenic variants in the gene, leading to dystrophin deficiency and progressive muscle degeneration. Thousands of variants with diverse types have been reported in , contributing to a broad clinical spectrum. While typically associated with severe phenotypes, pathogenic DMD variants may also cause Becker muscular dystrophy (BMD), a milder form with later-onset muscle weakness, or isolated dilated cardiomyopathy with minimal skeletal muscle involvement.
View Article and Find Full Text PDFACS Omega
August 2025
Department of Materials Engineering, Graduate School of Engineering, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8656, Japan.
Efficient delivery of oligonucleotide drugs to muscle tissues remains a significant challenge in nanomedicine and oligonucleotide therapeutics. A primary obstacle is the blood-muscle barrier, a continuous endothelium within muscle tissues that impedes the extravasation of conventional nanomedicines, typically ranging from a few tens of nanometers to 100 nm. To address this challenge, we developed an ultrasmall oligonucleotide nanomedicine, termed the unit polyion complex (uPIC), using a single molecular oligonucleotide with Y-shaped block catiomers.
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