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We report that BMD heart failure is associated with a significantly decreased number of cardiovascular progenitor cells, reduced cardiac fibroblast migration, and ex vivo survival.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Becker muscular dystrophy belongs to a class of genetically inherited dystrophin deficiencies. It affects male patients and results in progressive skeletal muscle degeneration and dilated cardiomyopathy leading to heart failure. It is a relatively mild form of dystrophin deficiency, which allows patients to be on a heart transplant list. In this unique situation, the explanted heart is a rare opportunity to study the degenerative process of dystrophin-deficient cardiac tissue. Heart tissue was excised, dissociated, and analyzed. The fractional content of c-kit\n                      <jats:sup>+<\/jats:sup>\n                      \/CD45\n                      <jats:sup>\u2212<\/jats:sup>\n                      cardiovascular progenitor cells (CVPCs) and cardiac fibroblast migration were compared to control samples of atrial tissue. Control tissue was obtained from the hearts of healthy organ donor\u2019s during heart transplantation procedures.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We report significantly decreased CVPCs (c-kit\n                      <jats:sup>+<\/jats:sup>\n                      \/CD45\n                      <jats:sup>\u2212<\/jats:sup>\n                      ) throughout the heart tissue of a BMD patient, and reduced numbers of phase-bright cells presenting c-kit positivity in the dystrophin-deficient cultured explants. In addition, ex vivo CVPCs survival and cardiac fibroblasts migration were significantly reduced, suggesting reduced homeostatic support and irreversible tissue remodeling.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Our findings associate genetically derived heart failure in a dystrophin-deficient patient with decreased c-kit\n                      <jats:sup>+<\/jats:sup>\n                      \/CD45\n                      <jats:sup>\u2212<\/jats:sup>\n                      CVPCs and their resilience, possibly hinting at a lack of cardioprotective capability and\/or reduced homeostatic support. This also correlates with reduced plasticity of the explanted cardiac tissue, related to the process of irreversible remodeling in the BMD patient\u2019s heart.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13023-019-1257-4","type":"journal-article","created":{"date-parts":[[2020,3,5]],"date-time":"2020-03-05T05:03:31Z","timestamp":1583384611000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Cardiovascular progenitor cells and tissue plasticity are reduced in a myocardium affected by Becker muscular dystrophy"],"prefix":"10.1186","volume":"15","author":[{"given":"Martin","family":"Pesl","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarka","family":"Jelinkova","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guido","family":"Caluori","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Holicka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan","family":"Krejci","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Petr","family":"Nemec","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aneta","family":"Kohutova","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vita","family":"Zampachova","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Petr","family":"Dvorak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vladimir","family":"Rotrekl","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2020,3,5]]},"reference":[{"key":"1257_CR1","doi-asserted-by":"publisher","first-page":"356","DOI":"10.4330\/wjc.v8.i6.356","volume":"8","author":"R Ho","year":"2016","unstructured":"Ho R, Nguyen M-L, Mather P. 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PLoS One. 2013;8:e67389.","journal-title":"PLoS One"}],"container-title":["Orphanet Journal of Rare Diseases"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13023-019-1257-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s13023-019-1257-4\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13023-019-1257-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,3,4]],"date-time":"2021-03-04T19:21:26Z","timestamp":1614885686000},"score":1,"resource":{"primary":{"URL":"https:\/\/ojrd.biomedcentral.com\/articles\/10.1186\/s13023-019-1257-4"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,3,5]]},"references-count":40,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["1257"],"URL":"https:\/\/doi.org\/10.1186\/s13023-019-1257-4","relation":{},"ISSN":["1750-1172"],"issn-type":[{"value":"1750-1172","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,3,5]]},"assertion":[{"value":"23 July 2019","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"19 November 2019","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"5 March 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"This study was carried out in accordance with the recommendations of the Masaryk University Ethics Committee and St. Ann\u2019s University Hospital Ethics Committee, issuing approval nr. 51\u2009V\/2015 - approved consent for heart tissue sampling and analysis, and genetic information analysis (DNA and circulating free DNA), protocol was approved on 12th August 2015. The \u201chealthy donor\u201d (HD) control samples were obtained from donors diagnosed with brain death. The hearts were brought to the transplantation center for transplantation and were provided for scientific analysis after the anonymization of donor data. Thus, individual information, such as names of donors, was not available to investigators. Samples from deceased donors were not further addressed by the ethics committee since the transplantation system is opt-out according to the law 285\/2002 Sb. in the Czech Republic. At the same time, due to anonymization, family members were not reachable.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"The study subject gave written informed consent regarding explanted organ sampling and publication of results.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"65"}}