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Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B

Two of the main pathologies characterizing dysferlinopathies are disrupted muscle membrane repair and chronic inflammation, which lead to symptoms of muscle weakness and wasting. Here, we used recombinant human Galectin-1 (rHsGal-1) as a therapeutic for LGMD2B mouse and human models. Various redox a...

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Autores principales: Vallecillo-Zúniga, Mary L., Poulson, Peter Daniel, Luddington, Jacob S., Arnold, Christian J., Rathgeber, Matthew, Kartchner, Braden C., Hayes, Spencer, Gill, Hailie, Valdoz, Jonard C., Spallino, Jonathan L., Garfield, Seth, Dodson, Ethan L., Arthur, Connie M., Stowell, Sean R., Van Ry, Pam M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8621416/
https://www.ncbi.nlm.nih.gov/pubmed/34831431
http://dx.doi.org/10.3390/cells10113210
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author Vallecillo-Zúniga, Mary L.
Poulson, Peter Daniel
Luddington, Jacob S.
Arnold, Christian J.
Rathgeber, Matthew
Kartchner, Braden C.
Hayes, Spencer
Gill, Hailie
Valdoz, Jonard C.
Spallino, Jonathan L.
Garfield, Seth
Dodson, Ethan L.
Arthur, Connie M.
Stowell, Sean R.
Van Ry, Pam M.
author_facet Vallecillo-Zúniga, Mary L.
Poulson, Peter Daniel
Luddington, Jacob S.
Arnold, Christian J.
Rathgeber, Matthew
Kartchner, Braden C.
Hayes, Spencer
Gill, Hailie
Valdoz, Jonard C.
Spallino, Jonathan L.
Garfield, Seth
Dodson, Ethan L.
Arthur, Connie M.
Stowell, Sean R.
Van Ry, Pam M.
author_sort Vallecillo-Zúniga, Mary L.
collection PubMed
description Two of the main pathologies characterizing dysferlinopathies are disrupted muscle membrane repair and chronic inflammation, which lead to symptoms of muscle weakness and wasting. Here, we used recombinant human Galectin-1 (rHsGal-1) as a therapeutic for LGMD2B mouse and human models. Various redox and multimerization states of Gal-1 show that rHsGal-1 is the most effective form in both increasing muscle repair and decreasing inflammation, due to its monomer-dimer equilibrium. Dose-response testing shows an effective 25-fold safety profile between 0.54 and 13.5 mg/kg rHsGal-1 in Bla/J mice. Mice treated weekly with rHsGal-1 showed downregulation of canonical NF-κB inflammation markers, decreased muscle fat deposition, upregulated anti-inflammatory cytokines, increased membrane repair, and increased functional movement compared to non-treated mice. Gal-1 treatment also resulted in a positive self-upregulation loop of increased endogenous Gal-1 expression independent of NF-κB activation. A similar reduction in disease pathologies in patient-derived human cells demonstrates the therapeutic potential of Gal-1 in LGMD2B patients.
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spelling pubmed-86214162021-11-27 Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B Vallecillo-Zúniga, Mary L. Poulson, Peter Daniel Luddington, Jacob S. Arnold, Christian J. Rathgeber, Matthew Kartchner, Braden C. Hayes, Spencer Gill, Hailie Valdoz, Jonard C. Spallino, Jonathan L. Garfield, Seth Dodson, Ethan L. Arthur, Connie M. Stowell, Sean R. Van Ry, Pam M. Cells Article Two of the main pathologies characterizing dysferlinopathies are disrupted muscle membrane repair and chronic inflammation, which lead to symptoms of muscle weakness and wasting. Here, we used recombinant human Galectin-1 (rHsGal-1) as a therapeutic for LGMD2B mouse and human models. Various redox and multimerization states of Gal-1 show that rHsGal-1 is the most effective form in both increasing muscle repair and decreasing inflammation, due to its monomer-dimer equilibrium. Dose-response testing shows an effective 25-fold safety profile between 0.54 and 13.5 mg/kg rHsGal-1 in Bla/J mice. Mice treated weekly with rHsGal-1 showed downregulation of canonical NF-κB inflammation markers, decreased muscle fat deposition, upregulated anti-inflammatory cytokines, increased membrane repair, and increased functional movement compared to non-treated mice. Gal-1 treatment also resulted in a positive self-upregulation loop of increased endogenous Gal-1 expression independent of NF-κB activation. A similar reduction in disease pathologies in patient-derived human cells demonstrates the therapeutic potential of Gal-1 in LGMD2B patients. MDPI 2021-11-17 /pmc/articles/PMC8621416/ /pubmed/34831431 http://dx.doi.org/10.3390/cells10113210 Text en © 2021 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Vallecillo-Zúniga, Mary L.
Poulson, Peter Daniel
Luddington, Jacob S.
Arnold, Christian J.
Rathgeber, Matthew
Kartchner, Braden C.
Hayes, Spencer
Gill, Hailie
Valdoz, Jonard C.
Spallino, Jonathan L.
Garfield, Seth
Dodson, Ethan L.
Arthur, Connie M.
Stowell, Sean R.
Van Ry, Pam M.
Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B
title Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B
title_full Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B
title_fullStr Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B
title_full_unstemmed Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B
title_short Therapeutic Benefit of Galectin-1: Beyond Membrane Repair, a Multifaceted Approach to LGMD2B
title_sort therapeutic benefit of galectin-1: beyond membrane repair, a multifaceted approach to lgmd2b
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8621416/
https://www.ncbi.nlm.nih.gov/pubmed/34831431
http://dx.doi.org/10.3390/cells10113210
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