{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,15]],"date-time":"2025-12-15T14:18:16Z","timestamp":1765808296000,"version":"3.37.3"},"reference-count":71,"publisher":"Oxford University Press (OUP)","issue":"10","license":[{"start":{"date-parts":[[2024,2,10]],"date-time":"2024-02-10T00:00:00Z","timestamp":1707523200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000069","name":"National Institute of Arthritis and Musculoskeletal and Skin Diseases","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000069","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"name":"University of Nevada, Reno School of Medicine"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,5,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Facioscapulohumeral muscular dystrophy (FSHD) is a progressive myopathy caused by the aberrant increased expression of the DUX4 retrogene in skeletal muscle cells. The DUX4 gene encodes a transcription factor that functions in zygotic genome activation and then is silenced in most adult somatic tissues. DUX4 expression in FSHD disrupts normal muscle cell function; however, the downstream pathogenic mechanisms are still unclear. Histologically, FSHD affected muscles show a characteristic dystrophic phenotype that is often accompanied by a pronounced immune cell infiltration, but the role of the immune system in FSHD is not understood. Previously, we used ACTA1;FLExDUX4 FSHD-like mouse models varying in severity as discovery tools to identify increased Interleukin 6 and microRNA-206 levels as serum biomarkers for FSHD disease severity. In this study, we use the ACTA1;FLExDUX4 chronic FSHD-like mouse model to provide insight into the immune response to DUX4 expression in skeletal muscles. We demonstrate that these FSHD-like muscles are enriched with the chemoattractant eotaxin and the cytotoxic eosinophil peroxidase, and exhibit muscle eosinophilia. We further identified muscle fibers with positive staining for eosinophil peroxidase in human FSHD muscle. Our data supports that skeletal muscle eosinophilia is a hallmark of FSHD pathology.<\/jats:p>","DOI":"10.1093\/hmg\/ddae019","type":"journal-article","created":{"date-parts":[[2024,2,10]],"date-time":"2024-02-10T12:32:22Z","timestamp":1707568342000},"page":"872-883","source":"Crossref","is-referenced-by-count":1,"title":["Muscle eosinophilia is a hallmark of chronic disease in facioscapulohumeral muscular dystrophy"],"prefix":"10.1093","volume":"33","author":[{"given":"Andreia M","family":"Nunes","sequence":"first","affiliation":[{"name":"Department of Pharmacology, University of Nevada, Reno School of Medicine , 1664 N. Virginia St., Reno, NV 89557 , United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Monique M","family":"Ramirez","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, University of Nevada, Reno School of Medicine , 1664 N. Virginia St., Reno, NV 89557 , United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Enrique","family":"Garcia-Collazo","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, University of Nevada, Reno School of Medicine , 1664 N. Virginia St., Reno, NV 89557 , United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takako Iida","family":"Jones","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, University of Nevada, Reno School of Medicine , 1664 N. Virginia St., Reno, NV 89557 , United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4283-1016","authenticated-orcid":false,"given":"Peter L","family":"Jones","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, University of Nevada, Reno School of Medicine , 1664 N. Virginia St., Reno, NV 89557 , United States"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2024,2,10]]},"reference":[{"key":"2024050512560641300_ref1","doi-asserted-by":"crossref","first-page":"S81","DOI":"10.1002\/mus.880181315","article-title":"Facioscapulohumeral muscular dystrophy in the dutch population","volume":"18","author":"Padberg","year":"1995","journal-title":"Muscle Nerve"},{"key":"2024050512560641300_ref2","first-page":"1","article-title":"Prevalence and incidence of rare diseases: bibliographic data","volume":"1","author":"Orphanet Report Series","year":"2022","journal-title":"Orphanet Report Series Rare diseases collection"},{"key":"2024050512560641300_ref3","doi-asserted-by":"crossref","first-page":"1056","DOI":"10.1212\/WNL.0000000000000797","article-title":"Population-based incidence and prevalence of facioscapulohumeral 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