Plasma lipidomic analysis shows a disease progression signature in mdx mice

Abstract Duchenne muscular dystrophy (DMD) is a rare genetic disorder affecting paediatric patients. The disease course is characterized by loss of muscle mass, which is rapidly substituted by fibrotic and adipose tissue. Clinical and preclinical models have clarified the processes leading to muscle...

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Autores principales:	Roula Tsonaka, Alexandre Seyer, Annemieke Aartsma-Rus, Pietro Spitali
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/bb7a18b1cea74e9bb5c40a2992c9ea8e
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spelling	oai:doaj.org-article:bb7a18b1cea74e9bb5c40a2992c9ea8e2021-12-02T16:07:04ZPlasma lipidomic analysis shows a disease progression signature in mdx mice10.1038/s41598-021-92406-62045-2322https://doaj.org/article/bb7a18b1cea74e9bb5c40a2992c9ea8e2021-06-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-92406-6https://doaj.org/toc/2045-2322Abstract Duchenne muscular dystrophy (DMD) is a rare genetic disorder affecting paediatric patients. The disease course is characterized by loss of muscle mass, which is rapidly substituted by fibrotic and adipose tissue. Clinical and preclinical models have clarified the processes leading to muscle damage and myofiber degeneration. Analysis of the fat component is however emerging as more evidence shows how muscle fat fraction is associated with patient performance and prognosis. In this article we aimed to study whether alterations exist in the composition of lipids in plasma samples obtained from mouse models. Analysis of plasma samples was performed in 4 mouse models of DMD and wild-type mice by LC–MS. Longitudinal samplings of individual mice covering an observational period of 7 months were obtained to cover the different phases of the disease. We report clear elevation of glycerolipids and glycerophospholipids families in dystrophic mice compared to healthy mice. Triacylglycerols were the strongest contributors to the signatures in mice. Annotation of individual lipids confirmed the elevation of lipids belonging to these families as strongest discriminants between healthy and dystrophic mice. A few sphingolipids (such as ganglioside GM2, sphingomyelin and ceramide), sterol lipids (such as cholesteryl oleate and cholesteryl arachidonate) and a fatty acyl (stearic acid) were also found to be affected in dystrophic mice. Analysis of serum and plasma samples show how several lipids are affected in dystrophic mice affected by muscular dystrophy. This study sets the basis to further investigations to understand how the lipid signature relates to the disease biology and muscle performance.Roula TsonakaAlexandre SeyerAnnemieke Aartsma-RusPietro SpitaliNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-9 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Roula Tsonaka Alexandre Seyer Annemieke Aartsma-Rus Pietro Spitali Plasma lipidomic analysis shows a disease progression signature in mdx mice
description	Abstract Duchenne muscular dystrophy (DMD) is a rare genetic disorder affecting paediatric patients. The disease course is characterized by loss of muscle mass, which is rapidly substituted by fibrotic and adipose tissue. Clinical and preclinical models have clarified the processes leading to muscle damage and myofiber degeneration. Analysis of the fat component is however emerging as more evidence shows how muscle fat fraction is associated with patient performance and prognosis. In this article we aimed to study whether alterations exist in the composition of lipids in plasma samples obtained from mouse models. Analysis of plasma samples was performed in 4 mouse models of DMD and wild-type mice by LC–MS. Longitudinal samplings of individual mice covering an observational period of 7 months were obtained to cover the different phases of the disease. We report clear elevation of glycerolipids and glycerophospholipids families in dystrophic mice compared to healthy mice. Triacylglycerols were the strongest contributors to the signatures in mice. Annotation of individual lipids confirmed the elevation of lipids belonging to these families as strongest discriminants between healthy and dystrophic mice. A few sphingolipids (such as ganglioside GM2, sphingomyelin and ceramide), sterol lipids (such as cholesteryl oleate and cholesteryl arachidonate) and a fatty acyl (stearic acid) were also found to be affected in dystrophic mice. Analysis of serum and plasma samples show how several lipids are affected in dystrophic mice affected by muscular dystrophy. This study sets the basis to further investigations to understand how the lipid signature relates to the disease biology and muscle performance.
format	article
author	Roula Tsonaka Alexandre Seyer Annemieke Aartsma-Rus Pietro Spitali
author_facet	Roula Tsonaka Alexandre Seyer Annemieke Aartsma-Rus Pietro Spitali
author_sort	Roula Tsonaka
title	Plasma lipidomic analysis shows a disease progression signature in mdx mice
title_short	Plasma lipidomic analysis shows a disease progression signature in mdx mice
title_full	Plasma lipidomic analysis shows a disease progression signature in mdx mice
title_fullStr	Plasma lipidomic analysis shows a disease progression signature in mdx mice
title_full_unstemmed	Plasma lipidomic analysis shows a disease progression signature in mdx mice
title_sort	plasma lipidomic analysis shows a disease progression signature in mdx mice
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/bb7a18b1cea74e9bb5c40a2992c9ea8e
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Plasma lipidomic analysis shows a disease progression signature in mdx mice

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