Literature DB >> 33322031

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

Emma Rybalka1,2, Cara A Timpani1,2, Danielle A Debruin1,2, Ryan M Bagaric1,2, Dean G Campelj1,2, Alan Hayes1,2,3.   

Abstract

Myostatin inhibition therapy has held much promise for the treatment of muscle wasting disorders. This is particularly true for the fatal myopathy, Duchenne Muscular Dystrophy (DMD). Following on from promising pre-clinical data in dystrophin-deficient mice and dogs, several clinical trials were initiated in DMD patients using different modality myostatin inhibition therapies. All failed to show modification of disease course as dictated by the primary and secondary outcome measures selected: the myostatin inhibition story, thus far, is a failed clinical story. These trials have recently been extensively reviewed and reasons why pre-clinical data collected in animal models have failed to translate into clinical benefit to patients have been purported. However, the biological mechanisms underlying translational failure need to be examined to ensure future myostatin inhibitor development endeavors do not meet with the same fate. Here, we explore the biology which could explain the failed translation of myostatin inhibitors in the treatment of DMD.

Entities:  

Keywords:  Duchenne Muscular Dystrophy; clinical trials; muscle development; myostatin inhibition; skeletal muscle; translation

Year:  2020        PMID: 33322031      PMCID: PMC7764137          DOI: 10.3390/cells9122657

Source DB:  PubMed          Journal:  Cells        ISSN: 2073-4409            Impact factor:   6.600


  124 in total

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Journal:  PLoS One       Date:  2020-03-11       Impact factor: 3.240

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Journal:  Int J Mol Sci       Date:  2022-04-30       Impact factor: 6.208

Review 2.  Molecular Mechanisms and Current Treatment Options for Cancer Cachexia.

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Review 3.  CRISPR technologies for the treatment of Duchenne muscular dystrophy.

Authors:  Eunyoung Choi; Taeyoung Koo
Journal:  Mol Ther       Date:  2021-04-03       Impact factor: 11.454

Review 4.  Targeting the Activin Receptor Signaling to Counteract the Multi-Systemic Complications of Cancer and Its Treatments.

Authors:  Juha J Hulmi; Tuuli A Nissinen; Fabio Penna; Andrea Bonetto
Journal:  Cells       Date:  2021-02-28       Impact factor: 6.600

Review 5.  Deciphering Myostatin's Regulatory, Metabolic, and Developmental Influence in Skeletal Diseases.

Authors:  Catherine L Omosule; Charlotte L Phillips
Journal:  Front Genet       Date:  2021-03-29       Impact factor: 4.599

Review 6.  Common Pathogenic Mechanisms in Centronuclear and Myotubular Myopathies and Latest Treatment Advances.

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Review 7.  Neuromuscular Development and Disease: Learning From in vitro and in vivo Models.

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8.  Inactivation of Sirt6 ameliorates muscular dystrophy in mdx mice by releasing suppression of utrophin expression.

Authors:  Angelina M Georgieva; Xinyue Guo; Marek Bartkuhn; Stefan Günther; Carsten Künne; Christian Smolka; Ann Atzberger; Ulrich Gärtner; Kamel Mamchaoui; Eva Bober; Yonggang Zhou; Xuejun Yuan; Thomas Braun
Journal:  Nat Commun       Date:  2022-07-20       Impact factor: 17.694

Review 9.  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

  9 in total

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