{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"institution":[{"name":"bioRxiv"}],"indexed":{"date-parts":[[2026,4,11]],"date-time":"2026-04-11T08:03:57Z","timestamp":1775894637584,"version":"3.50.1"},"posted":{"date-parts":[[2025,2,1]]},"group-title":"Genetics","reference-count":56,"publisher":"openRxiv","license":[{"start":{"date-parts":[[2025,2,1]],"date-time":"2025-02-01T00:00:00Z","timestamp":1738368000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2025,2,9]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                <jats:p>\n                  Duchenne muscular dystrophy (DMD) patients suffer from skeletal and cardiopulmonary weakness, and interestingly up to one third are diagnosed on the autism spectrum. Dystrophin is an essential protein for regulating the transmission of intracellular force to the extracellular matrix within the skeletal muscle, but also plays key roles in neurobehavior and cognitive function. The mouse dystrophin gene (also abbreviated\n                  <jats:italic>Dmd<\/jats:italic>\n                  ) is X-linked and has several isoforms with tissue-specific expression, including the large\n                  <jats:italic>Dp427m<\/jats:italic>\n                  muscle transcript found in heart and skeletal muscle, and the\n                  <jats:italic>Dp427c<\/jats:italic>\n                  transcript that encodes the brain-specific dystrophin cerebellar protein. Understanding the functional requirements and pathways that are affected by dystrophin loss will impact dystrophin replacement gene therapy and exon-skipping correction strategies. We generated conditional\n                  <jats:italic>Dystrophin<\/jats:italic>\n                  knockout mice by targeting exon 52 of the mouse\n                  <jats:italic>Dystrophin<\/jats:italic>\n                  (\n                  <jats:italic>Dmd<\/jats:italic>\n                  <jats:sup>flox52<\/jats:sup>\n                  ) locus. We generated dystrophin constitutive and inducible myofiber knockout (\n                  <jats:italic>Dmd<\/jats:italic>\n                  mKO) mice to evaluate the tissue-specific function of the large skeletal muscle dystrophin isoform. Constitutive embryonic deletion of the\n                  <jats:italic>Dystrophin<\/jats:italic>\n                  gene exclusively in skeletal myofibers resulted in a severe skeletal muscle myopathy, dystrophic histopathology, and functional deficits compared to the\n                  <jats:italic>mdx<\/jats:italic>\n                  mouse. Transcriptomic analysis of skeletal myofibers of the\n                  <jats:italic>Dmd<\/jats:italic>\n                  mKO mice revealed the dysregulation of key extracellular matrix and cytokine signaling pathways. Separately, we generated Purkinje neuron cerebellar dystrophin knockout (\n                  <jats:italic>Dmd<\/jats:italic>\n                  :Pcp2 KO) mice that displayed neurobehavioral deficits in social approach, social memory, and spatial navigation and working memory. These studies reveal the essential requirement for dystrophin expression in both the skeletal muscle and brain for normal physiological and neurobehavioral function.\n                <\/jats:p>\n                <jats:sec>\n                  <jats:title>Significance Statement<\/jats:title>\n                  <jats:p>\n                    Duchenne muscular dystrophy is caused by the lack of a functional dystrophin protein in muscle. The large dystrophin (Dp427m) isoform is expressed in skeletal, cardiac, and smooth muscle, but its tissue-specific requirements remain unknown. We generated and characterized a conditional skeletal muscle knockout mouse (\n                    <jats:italic>Dmd<\/jats:italic>\n                    mKO). Constitutive embryonic genetic ablation of skeletal muscle\n                    <jats:italic>Dystrophin<\/jats:italic>\n                    resulted in muscle histopathologies similar to the\n                    <jats:italic>mdx<\/jats:italic>\n                    mouse, while postnatal muscle\n                    <jats:italic>Dystrophin<\/jats:italic>\n                    ablation resulted in milder pathologies. Ablating cerebellar\n                    <jats:italic>Dystrophin<\/jats:italic>\n                    Dp427c using a\n                    <jats:italic>Pcp2\/L7<\/jats:italic>\n                    -Cre driver resulted in sociobehavioral defects. Transcriptomic analysis of the\n                    <jats:italic>Dmd<\/jats:italic>\n                    mKO mice showed a severe reduction of extracellular matrix and cytokine signaling pathways. Our study reveals an essential role for skeletal muscle dystrophin and identifies essential pathways for modulation using dystrophin-replacement therapies.\n                  <\/jats:p>\n                <\/jats:sec>","DOI":"10.1101\/2025.01.30.635777","type":"posted-content","created":{"date-parts":[[2025,2,1]],"date-time":"2025-02-01T05:40:14Z","timestamp":1738388414000},"source":"Crossref","is-referenced-by-count":1,"title":["Conditional\n                  <i>Dystrophin<\/i>\n                  ablation in the skeletal muscle and brain causes profound effects on muscle function, neurobehavior, and extracellular matrix pathways"],"prefix":"10.64898","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0034-9950","authenticated-orcid":false,"given":"Muthukumar","family":"Karuppasamy","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Division of Neurology at the University of Alabama at Birmingham and Children\u2019s of Alabama, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2757-5157","authenticated-orcid":false,"given":"Katherine G.","family":"English","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Neurology at the University of Alabama at Birmingham and Children\u2019s of Alabama, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8886-1475","authenticated-orcid":false,"given":"James R.","family":"Conner","sequence":"additional","affiliation":[{"name":"Division of Genetics and Genomics, Boston Children\u2019s Hospital, Boston, MA 02115, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-5640-9669","authenticated-orcid":false,"given":"Shelby N.","family":"Rorrer","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Neurology at the University of Alabama at Birmingham and Children\u2019s of Alabama, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3029-0761","authenticated-orcid":false,"given":"Michael A.","family":"Lopez","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Neurology at the University of Alabama at Birmingham and Children\u2019s of Alabama, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0981-169X","authenticated-orcid":false,"given":"David K.","family":"Crossman","sequence":"additional","affiliation":[{"name":"Department of Genetics at the University of Alabama at Birmingham, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5409-5944","authenticated-orcid":false,"given":"Jodi R.","family":"Paul","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Neurobiology, University of Alabama at Birmingham, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8601-4080","authenticated-orcid":false,"given":"Miguel A.","family":"Monreal-Gutierrez","sequence":"additional","affiliation":[{"name":"Department of Physiology and Aging, University of Florida, Gainesville, FL 32610, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3813-8577","authenticated-orcid":false,"given":"Karen L.","family":"Gamble","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Neurobiology, University of Alabama at Birmingham, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5791-1441","authenticated-orcid":false,"given":"Karyn A.","family":"Esser","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Neurobiology, University of Alabama at Birmingham, Birmingham, AL 35294"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5597-1141","authenticated-orcid":false,"given":"Jeffrey J.","family":"Widrick","sequence":"additional","affiliation":[{"name":"Division of Genetics and Genomics, Boston Children\u2019s Hospital, Boston, MA 02115, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6126-0242","authenticated-orcid":false,"given":"Louis M.","family":"Kunkel","sequence":"additional","affiliation":[{"name":"Division of Genetics and Genomics, Boston Children\u2019s Hospital, Boston, MA 02115, USA"},{"name":"Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA"},{"name":"The Stem Cell Program, Boston Children\u2019s Hospital, Boston, MA 02115, USA"},{"name":"Harvard Stem Cell Institute, Cambridge, MA 02138, USA"},{"name":"The Manton Center for Orphan Disease Research at Boston Children\u2019s Hospital, Boston, MA 02115, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7406-5171","authenticated-orcid":false,"given":"Matthew S.","family":"Alexander","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Neurology at the University of Alabama at Birmingham and Children\u2019s of Alabama, Birmingham, AL 35294"},{"name":"UAB Center for Exercise Medicine at the University of Alabama at Birmingham, Birmingham, AL, 35294"},{"name":"UAB Civitan International Research Center (CIRC), at the University of Alabama at Birmingham, Birmingham, AL 35233"},{"name":"UAB Center for Neurodegeneration and Experimental Therapeutics (CNET), Birmingham, AL 35294, 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