ArticleFASEB journal : official publication of the Federation of American Societies for Experimental Biology2013
Selenoprotein N deficiency in mice is associated with abnormal lung development.
Article in FASEB journal : official publication of the Federation of American Societies for Experimental Biology, 2013. 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, 41 citations in OpenAlex.
- Selenoprotein N and SEPN1-Related Myopathies: Mechanisms, Models, and Therapeutic Perspectives.Biomolecules · 2026Review
- Zebrafish and cellular models of SELENON-Congenital myopathy exhibit novel embryonic and metabolic phenotypes.Skeletal muscle · 2025Article
- Impaired Upper Airway Muscle Function with Excessive or Deficient Dietary Intake of Selenium in Rats.Antioxidants (Basel, Switzerland) · 2024Article
- Oxidative Stress Induced by Selenium Deficiency Contributes to Inflammation, Apoptosis and Necroptosis in the Lungs of Calves.Antioxidants (Basel, Switzerland) · 2023Article
- Neonatal Selenium Deficiency Decreases Selenoproteins in the Lung and Impairs Pulmonary Alveolar Development.Antioxidants (Basel, Switzerland) · 2022Article
- NovelFrontiers in genetics · 2022Article
- Beyond antioxidants: Selenium and skeletal muscle mitochondria.Frontiers in veterinary science · 2022Review
- Review
- Tolerance to Selenoprotein Loss Differs between Human and Mouse.Molecular biology and evolution · 2020Article
- A maladaptive ER stress response triggers dysfunction in highly active muscles of mice with SELENON loss.Redox biology · 2019Article
- Damage to the myogenic differentiation of C2C12 cells by heat stress is associated with up-regulation of several selenoproteins.Scientific reports · 2018Article
- Myostatin inhibition using mRK35 produces skeletal muscle growth and tubular aggregate formation in wild type and TgACTA1D286G nemaline myopathy mice.Human molecular genetics · 2018Article
- Early Onset of Sleep-Disordered Breathing in Two Children WithJournal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2017Article
- Endoplasmic Reticulum Oxidative Stress Triggers Tgf-Beta-Dependent Muscle Dysfunction by Accelerating Ascorbic Acid Turnover.Scientific reports · 2017Article
- A Chronic Obstructive Pulmonary Disease Susceptibility Gene, FAM13A, Regulates Protein Stability of β-Catenin.American journal of respiratory and critical care medicine · 2016Article
- Treatment with ActRIIB-mFc Produces Myofiber Growth and Improves Lifespan in the Acta1 H40Y Murine Model of Nemaline Myopathy.The American journal of pathology · 2016Article
- Dietary selenium augments sarcoplasmic calcium release and mechanical performance in mice.Nutrition & metabolism · 2016Article
- Automated measurement of pulmonary emphysema and small airway remodeling in cigarette smoke-exposed mice.Journal of visualized experiments : JoVE · 2015Article
- Haploinsufficiency of Hedgehog interacting protein causes increased emphysema induced by cigarette smoke through network rewiring.Genome medicine · 2015Article
- Respiratory assessment in centronuclear myopathies.Muscle & nerve · 2014Review
Corrections and comments
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Authors and funding
9 authors at 7 institutions in 1 country.
Funding
Abstract
Mutations in the human SEPN1 gene, encoding selenoprotein N (SepN), cause SEPN1-related myopathy (SEPN1-RM) characterized by muscle weakness, spinal rigidity, and respiratory insufficiency. As with other members of the selenoprotein family, selenoprotein N incorporates selenium in the form of selenocysteine (Sec). Most selenoproteins that have been functionally characterized are involved in oxidation-reduction (redox) reactions, with the Sec residue located at their catalytic site. To model SEPN1-RM, we generated a Sepn1-knockout (Sepn1(-/-)) mouse line. Homozygous Sepn1(-/-) mice are fertile, and their weight and lifespan are comparable to wild-type (WT) animals. Under baseline conditions, the muscle histology of Sepn1(-/-) mice remains normal, but subtle core lesions could be detected in skeletal muscle after inducing oxidative stress. Ryanodine receptor (RyR) calcium release channels showed lower sensitivity to caffeine in SepN deficient myofibers, suggesting a possible role of SepN in RyR regulation. SepN deficiency also leads to abnormal lung development characterized by enlarged alveoli, which is associated with decreased tissue elastance and increased quasi-static compliance of Sepn1(-/-) lungs. This finding raises the possibility that the respiratory syndrome observed in patients with SEPN1 mutations may have a primary pulmonary component in addition to the weakness of respiratory muscles.
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