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Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis

Duchenne muscular dystrophy (DMD) is a degenerative muscle disorder characterized by a lack of dystrophin expression in the sarcolemma of muscle fibers. DMD patients acquire bone abnormalities including osteopenia, fragility fractures, and scoliosis indicating a deficiency in skeletal homeostasis. T...

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Autores principales: Li, Hongshuai, Lu, Aiping, Gao, Xueqin, Tang, Ying, Ravuri, Sudheer, Wang, Bing, Huard, Johnny
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8073674/
https://www.ncbi.nlm.nih.gov/pubmed/33923553
http://dx.doi.org/10.3390/metabo11040247
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author Li, Hongshuai
Lu, Aiping
Gao, Xueqin
Tang, Ying
Ravuri, Sudheer
Wang, Bing
Huard, Johnny
author_facet Li, Hongshuai
Lu, Aiping
Gao, Xueqin
Tang, Ying
Ravuri, Sudheer
Wang, Bing
Huard, Johnny
author_sort Li, Hongshuai
collection PubMed
description Duchenne muscular dystrophy (DMD) is a degenerative muscle disorder characterized by a lack of dystrophin expression in the sarcolemma of muscle fibers. DMD patients acquire bone abnormalities including osteopenia, fragility fractures, and scoliosis indicating a deficiency in skeletal homeostasis. The dKO (dystrophin/Utrophin double knockout) is a more severe mouse model of DMD than the mdx mouse (dystrophin deficient), and display numerous clinically-relevant manifestations, including a spectrum of degenerative changes outside skeletal muscle including bone, articular cartilage, and intervertebral discs. To examine the influence of systemic factors on the bone abnormalities and healing in DMD, parabiotic pairing between dKO mice and mdx mice was established. Notably, heterochronic parabiosis with young mdx mice significantly increased bone mass and improved bone micro-structure in old dKO-hetero mice, which showed progressive bone deterioration. Furthermore, heterochronic parabiosis with WT C56/10J mice significantly improved tibia bone defect healing in dKO-homo mice. These results suggest that systemic blood-borne factor(s) and/or progenitors from WT and young mdx mice can influence the bone deficiencies in dKO mice. Understanding these circulating factors or progenitor cells that are responsible to alleviate the bone abnormalities in dKO mice after heterochronic parabiosis might be useful for the management of poor bone health in DMD.
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spelling pubmed-80736742021-04-27 Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis Li, Hongshuai Lu, Aiping Gao, Xueqin Tang, Ying Ravuri, Sudheer Wang, Bing Huard, Johnny Metabolites Article Duchenne muscular dystrophy (DMD) is a degenerative muscle disorder characterized by a lack of dystrophin expression in the sarcolemma of muscle fibers. DMD patients acquire bone abnormalities including osteopenia, fragility fractures, and scoliosis indicating a deficiency in skeletal homeostasis. The dKO (dystrophin/Utrophin double knockout) is a more severe mouse model of DMD than the mdx mouse (dystrophin deficient), and display numerous clinically-relevant manifestations, including a spectrum of degenerative changes outside skeletal muscle including bone, articular cartilage, and intervertebral discs. To examine the influence of systemic factors on the bone abnormalities and healing in DMD, parabiotic pairing between dKO mice and mdx mice was established. Notably, heterochronic parabiosis with young mdx mice significantly increased bone mass and improved bone micro-structure in old dKO-hetero mice, which showed progressive bone deterioration. Furthermore, heterochronic parabiosis with WT C56/10J mice significantly improved tibia bone defect healing in dKO-homo mice. These results suggest that systemic blood-borne factor(s) and/or progenitors from WT and young mdx mice can influence the bone deficiencies in dKO mice. Understanding these circulating factors or progenitor cells that are responsible to alleviate the bone abnormalities in dKO mice after heterochronic parabiosis might be useful for the management of poor bone health in DMD. MDPI 2021-04-16 /pmc/articles/PMC8073674/ /pubmed/33923553 http://dx.doi.org/10.3390/metabo11040247 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
Li, Hongshuai
Lu, Aiping
Gao, Xueqin
Tang, Ying
Ravuri, Sudheer
Wang, Bing
Huard, Johnny
Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis
title Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis
title_full Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis
title_fullStr Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis
title_full_unstemmed Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis
title_short Improved Bone Quality and Bone Healing of Dystrophic Mice by Parabiosis
title_sort improved bone quality and bone healing of dystrophic mice by parabiosis
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8073674/
https://www.ncbi.nlm.nih.gov/pubmed/33923553
http://dx.doi.org/10.3390/metabo11040247
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