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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\u2013MS. Longitudinal samplings of individual mice covering an observational period of 7\u00a0months 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.<\/jats:p>","DOI":"10.1038\/s41598-021-92406-6","type":"journal-article","created":{"date-parts":[[2021,6,21]],"date-time":"2021-06-21T10:03:10Z","timestamp":1624269790000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["Plasma lipidomic analysis shows a disease progression signature in mdx mice"],"prefix":"10.1038","volume":"11","author":[{"given":"Roula","family":"Tsonaka","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandre","family":"Seyer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annemieke","family":"Aartsma-Rus","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pietro","family":"Spitali","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,6,21]]},"reference":[{"key":"92406_CR1","doi-asserted-by":"publisher","first-page":"845","DOI":"10.1016\/S0140-6736(12)61897-2","volume":"381","author":"E Mercuri","year":"2013","unstructured":"Mercuri, E. & Muntoni, F. 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LUMC has patents on exon skipping, which were licensed to BioMarin and subsequently sublicensed to Sarepta. As co-inventor of some of these patents, Dr. Aartsma-Rus is entitled to a share of royalties. Pietro Spitali has served as ad-hoc consultant for Esperare, Summit PLC, Biomarin, Solid Bio, Bioclinica B.V., Marathon Biopharma, Astellas, Epirium Bio and Mantis therapeutics. He has served as in the scientific advisory board of Esperare, Epirium Bio and Summit PLC. Remuneration for these activities is paid to LUMC. A.A.-R. is also an ad hoc consultant for PTC Therapeutics, Sarepta Therapeutics, CRISPR Therapeutics, Summit PLC, Alpha Anomeric, BioMarin Pharmaceuticals, Eisai, Astra Zeneca, Global Guidepoint and GLG consultancy, Grunenthal, Wave and BioClinica, having been a member of the Duchenne Network Steering Committee (BioMarin) and being a member of the scientific advisory boards of ProQR and Philae Pharmaceuticals. Remuneration for these activities is paid to LUMC. LUMC also received speaker honoraria from PTC Therapeutics and BioMarin Pharmaceuticals and funding for contract research from Italpharmaco and Alpha Anomeric. Alexandre Seyer reports being employed by MedDay and receives salary by MedDay.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"12993"}}