{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,4]],"date-time":"2026-06-04T14:31:08Z","timestamp":1780583468509,"version":"3.54.1"},"reference-count":98,"publisher":"The Company of Biologists","issue":"4","license":[{"start":{"date-parts":[[2024,5,3]],"date-time":"2024-05-03T00:00:00Z","timestamp":1714694400000},"content-version":"vor","delay-in-days":32,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"crossref","award":["GRNT1144772"],"award-info":[{"award-number":["GRNT1144772"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001782","name":"University of Melbourne","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001782","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,4,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Duchenne muscular dystrophy (DMD) is a devastating monogenic skeletal muscle-wasting disorder. Although many pharmacological and genetic interventions have been reported in preclinical studies, few have progressed to clinical trials with meaningful benefit. Identifying therapeutic potential can be limited by availability of suitable preclinical mouse models. More rigorous testing across models with varied background strains and mutations can identify treatments for clinical success. Here, we report the generation of a DMD mouse model with a CRISPR-induced deletion within exon 62 of the dystrophin gene (Dmd) and the first generated in BALB\/c mice. Analysis of mice at 3, 6 and 12\u2005months of age confirmed loss of expression of the dystrophin protein isoform Dp427 and resultant dystrophic pathology in limb muscles and the diaphragm, with evidence of centrally nucleated fibers, increased inflammatory markers and fibrosis, progressive decline in muscle function, and compromised trabecular bone development. The BALB\/c.mdx62 mouse is a novel model of DMD with associated variations in the immune response and muscle phenotype, compared with those of existing models. It represents an important addition to the preclinical model toolbox for developing therapeutic strategies.<\/jats:p>","DOI":"10.1242\/dmm.050502","type":"journal-article","created":{"date-parts":[[2024,4,11]],"date-time":"2024-04-11T08:11:44Z","timestamp":1712823104000},"source":"Crossref","is-referenced-by-count":2,"title":["The BALB\/c.<i>mdx<\/i>62 mouse exhibits a dystrophic muscle pathology and is a model of Duchenne muscular dystrophy"],"prefix":"10.1242","volume":"17","author":[{"given":"Kristy","family":"Swiderski","sequence":"first","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Audrey S.","family":"Chan","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marco J.","family":"Herold","sequence":"additional","affiliation":[{"name":"The Walter and Eliza Hall Institute of Medical Research 2 , Parkville, VIC 3052 , Australia"},{"name":"The University of Melbourne 3 Department of Medical Biology , , Melbourne, VIC 3052 , Australia"},{"name":"Olivia Newton-John Cancer Research Institute 4 , Heidelberg, VIC 3084 , Australia"},{"name":"School of Cancer Medicine, La Trobe University 5 , Heidelberg, VIC 3084 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew J.","family":"Kueh","sequence":"additional","affiliation":[{"name":"The Walter and Eliza Hall Institute of Medical Research 2 , Parkville, VIC 3052 , Australia"},{"name":"The University of Melbourne 3 Department of Medical Biology , , Melbourne, VIC 3052 , Australia"},{"name":"Olivia Newton-John Cancer Research Institute 4 , Heidelberg, VIC 3084 , Australia"},{"name":"School of Cancer Medicine, La Trobe University 5 , Heidelberg, VIC 3084 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jin D.","family":"Chung","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin P.","family":"Hardee","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer","family":"Trieu","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annabel","family":"Chee","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Timur","family":"Naim","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Gregorevic","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9220-9810","authenticated-orcid":false,"given":"Gordon S.","family":"Lynch","sequence":"additional","affiliation":[{"name":"Centre for Muscle Research 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"},{"name":"The University of Melbourne 1 , Department of Anatomy and Physiology , , Melbourne, VIC 3010 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,5,3]]},"reference":[{"key":"2024050307145402100_DMM050502C1","doi-asserted-by":"publisher","first-page":"421","DOI":"10.1016\/j.ccr.2005.10.004","article-title":"Dystrophin glycoprotein complex dysfunction: a regulatory link between muscular dystrophy and cancer cachexia","volume":"8","author":"Acharyya","year":"2005","journal-title":"Cancer Cell"},{"key":"2024050307145402100_DMM050502C2","doi-asserted-by":"publisher","first-page":"eaan8081","DOI":"10.1126\/scitranslmed.aan8081","article-title":"Single-cut genome editing restores dystrophin expression in a new mouse model of muscular dystrophy","volume":"9","author":"AmoasII","year":"2017","journal-title":"Sci. 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