Literature DB >> 31830002

Glucocorticoids counteract hypertrophic effects of myostatin inhibition in dystrophic muscle.

David W Hammers1,2, Cora C Hart1,2, Andreas Patsalos3,4,5, Michael K Matheny1,2, Lillian A Wright1,2, Laszlo Nagy3,4,5, H Lee Sweeney1,2.   

Abstract

Duchenne muscular dystrophy (DMD) is a devastating genetic muscle disease resulting in progressive muscle degeneration and wasting. Glucocorticoids, specifically prednisone/prednisolone and deflazacort, are commonly used by DMD patients. Emerging DMD therapeutics include those targeting the muscle-wasting factor, myostatin (Mstn). The aim of this study was to investigate how chronic glucocorticoid treatment impacts the efficacy of Mstn inhibition in the D2.mdx mouse model of DMD. We report that chronic treatment of dystrophic mice with prednisolone (Pred) causes significant muscle wasting, entailing both activation of the ubiquitin-proteasome degradation pathway and inhibition of muscle protein synthesis. Combining Pred with Mstn inhibition, using a modified Mstn propeptide (dnMstn), completely abrogates the muscle hypertrophic effects of Mstn inhibition independently of Mstn expression or SMAD3 activation. Transcriptomic analysis identified that combining Pred with dnMstn treatment affects gene expression profiles associated with inflammation, metabolism, and fibrosis. Additionally, we demonstrate that Pred-induced muscle atrophy is not prevented by Mstn ablation. Therefore, glucocorticoids interfere with potential muscle mass benefits associated with targeting Mstn, and the ramifications of glucocorticoid use should be a consideration during clinical trial design for DMD therapeutics. These results have significant implications for past and future Mstn inhibition trials in DMD.

Entities:  

Keywords:  Monogenic diseases; Muscle; Muscle Biology; Therapeutics; Translation

Mesh:

Substances:

Year:  2020        PMID: 31830002      PMCID: PMC7030817          DOI: 10.1172/jci.insight.133276

Source DB:  PubMed          Journal:  JCI Insight        ISSN: 2379-3708


  73 in total

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Authors:  Chengzu Long; Leonela Amoasii; Alex A Mireault; John R McAnally; Hui Li; Efrain Sanchez-Ortiz; Samadrita Bhattacharyya; John M Shelton; Rhonda Bassel-Duby; Eric N Olson
Journal:  Science       Date:  2015-12-31       Impact factor: 47.728

2.  Stain-Free total protein staining is a superior loading control to β-actin for Western blots.

Authors:  Jennifer E Gilda; Aldrin V Gomes
Journal:  Anal Biochem       Date:  2013-06-06       Impact factor: 3.365

3.  Myostatin inhibitor ACE-031 treatment of ambulatory boys with Duchenne muscular dystrophy: Results of a randomized, placebo-controlled clinical trial.

Authors:  Craig Campbell; Hugh J McMillan; Jean K Mah; Mark Tarnopolsky; Kathryn Selby; Ty McClure; Dawn M Wilson; Matthew L Sherman; Diana Escolar; Kenneth M Attie
Journal:  Muscle Nerve       Date:  2016-12-23       Impact factor: 3.217

4.  Effect of genetic background on the dystrophic phenotype in mdx mice.

Authors:  William D Coley; Laurent Bogdanik; Maria Candida Vila; Qing Yu; Jack H Van Der Meulen; Sree Rayavarapu; James S Novak; Marie Nearing; James L Quinn; Allison Saunders; Connor Dolan; Whitney Andrews; Catherine Lammert; Andrew Austin; Terence A Partridge; Gregory A Cox; Cathleen Lutz; Kanneboyina Nagaraju
Journal:  Hum Mol Genet       Date:  2015-11-12       Impact factor: 6.150

5.  Thigh muscle mass and function in patients treated with glucocorticoids.

Authors:  F F Horber; J R Scheidegger; B E Grünig; F J Frey
Journal:  Eur J Clin Invest       Date:  1985-12       Impact factor: 4.686

6.  Randomized, double-blind six-month trial of prednisone in Duchenne's muscular dystrophy.

Authors:  J R Mendell; R T Moxley; R C Griggs; M H Brooke; G M Fenichel; J P Miller; W King; L Signore; S Pandya; J Florence
Journal:  N Engl J Med       Date:  1989-06-15       Impact factor: 91.245

7.  Supraphysiological levels of GDF11 induce striated muscle atrophy.

Authors:  David W Hammers; Melissa Merscham-Banda; Jennifer Ying Hsiao; Stefan Engst; James J Hartman; H Lee Sweeney
Journal:  EMBO Mol Med       Date:  2017-04       Impact factor: 12.137

8.  Osteopontin ablation ameliorates muscular dystrophy by shifting macrophages to a pro-regenerative phenotype.

Authors:  Joana Capote; Irina Kramerova; Leonel Martinez; Sylvia Vetrone; Elisabeth R Barton; H Lee Sweeney; M Carrie Miceli; Melissa J Spencer
Journal:  J Cell Biol       Date:  2016-04-18       Impact factor: 10.539

9.  Blocking extracellular activation of myostatin as a strategy for treating muscle wasting.

Authors:  M Pirruccello-Straub; J Jackson; S Wawersik; M T Webster; L Salta; K Long; W McConaughy; A Capili; C Boston; G J Carven; N K Mahanthappa; K J Turner; A Donovan
Journal:  Sci Rep       Date:  2018-02-02       Impact factor: 4.379

10.  Phase 1 Study of Edasalonexent (CAT-1004), an Oral NF-κB Inhibitor, in Pediatric Patients with Duchenne Muscular Dystrophy.

Authors:  Erika Finanger; Krista Vandenborne; Richard S Finkel; H Lee Sweeney; Gihan Tennekoon; Sabrina Yum; Maria Mancini; Pradeep Bista; Andrew Nichols; Hanlan Liu; Angelika Fretzen; Joanne M Donovan
Journal:  J Neuromuscul Dis       Date:  2019
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  6 in total

1.  The Effect of Deflazacort Treatment on the Functioning of Skeletal Muscle Mitochondria in Duchenne Muscular Dystrophy.

Authors:  Mikhail V Dubinin; Eugeny Yu Talanov; Kirill S Tenkov; Vlada S Starinets; Natalia V Belosludtseva; Konstantin N Belosludtsev
Journal:  Int J Mol Sci       Date:  2020-11-19       Impact factor: 5.923

2.  The D2.mdx mouse as a preclinical model of the skeletal muscle pathology associated with Duchenne muscular dystrophy.

Authors:  David W Hammers; Cora C Hart; Michael K Matheny; Lillian A Wright; Megan Armellini; Elisabeth R Barton; H Lee Sweeney
Journal:  Sci Rep       Date:  2020-08-21       Impact factor: 4.379

3.  The Failed Clinical Story of Myostatin Inhibitors against Duchenne Muscular Dystrophy: Exploring the Biology behind the Battle.

Authors:  Emma Rybalka; Cara A Timpani; Danielle A Debruin; Ryan M Bagaric; Dean G Campelj; Alan Hayes
Journal:  Cells       Date:  2020-12-10       Impact factor: 6.600

4.  Muscle Damage in Dystrophic mdx Mice Is Influenced by the Activity of Ca2+-Activated KCa3.1 Channels.

Authors:  Marta Morotti; Stefano Garofalo; Germana Cocozza; Fabrizio Antonangeli; Valeria Bianconi; Chiara Mozzetta; Maria Egle De Stefano; Riccardo Capitani; Heike Wulff; Cristina Limatola; Myriam Catalano; Francesca Grassi
Journal:  Life (Basel)       Date:  2022-04-05

Review 5.  CRISPR-Based Therapeutic Gene Editing for Duchenne Muscular Dystrophy: Advances, Challenges and Perspectives.

Authors:  Guofang Chen; Tingyi Wei; Hui Yang; Guoling Li; Haisen Li
Journal:  Cells       Date:  2022-09-22       Impact factor: 7.666

Review 6.  Antimyostatin Treatment in Health and Disease: The Story of Great Expectations and Limited Success.

Authors:  Tue L Nielsen; John Vissing; Thomas O Krag
Journal:  Cells       Date:  2021-03-03       Impact factor: 6.600

  6 in total

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