{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,9]],"date-time":"2026-03-09T21:13:26Z","timestamp":1773090806423,"version":"3.50.1"},"reference-count":39,"publisher":"Proceedings of the National Academy of Sciences","issue":"23","content-domain":{"domain":["www.pnas.org"],"crossmark-restriction":true},"short-container-title":["Proc. Natl. Acad. Sci. U.S.A."],"published-print":{"date-parts":[[2009,6,9]]},"abstract":"<jats:p>\n            Skeletal muscle is formed via fusion of myoblasts, a well-studied process in\n            <jats:italic>Drosophila<\/jats:italic>\n            . In vertebrates however, this process is less well understood, and whether there is evolutionary conservation with the proteins studied in flies is under investigation.\n            <jats:italic>Sticks and stones (Sns)<\/jats:italic>\n            , a cell surface protein found on\n            <jats:italic>Drosophila<\/jats:italic>\n            myoblasts, has structural homology to nephrin. Nephrin is a protein expressed in kidney that is part of the filtration barrier formed by podocytes. No previous study has established any role for nephrin in skeletal muscle. We show, using two models, zebrafish and mice, that the absence of nephrin results in poorly developed muscles and incompletely fused myotubes, respectively. Although nephrin-knockout (nephrin\n            <jats:italic>\n              <jats:sup>KO<\/jats:sup>\n            <\/jats:italic>\n            ) myoblasts exhibit prolonged activation of MAPK\/ERK pathway during myogenic differentiation, expression of myogenin does not seem to be altered. Nevertheless, MAPK pathway blockade does not rescue myoblast fusion. Co-cultures of unaffected human fetal myoblasts with nephrin\n            <jats:italic>\n              <jats:sup>KO<\/jats:sup>\n            <\/jats:italic>\n            myoblasts or myotubes restore the formation of mature myotubes; however, the contribution of nephrin\n            <jats:italic>\n              <jats:sup>KO<\/jats:sup>\n            <\/jats:italic>\n            myoblasts is minimal. These studies suggest that nephrin plays a role in secondary fusion of myoblasts into nascent myotubes, thus establishing a possible functional conservation with\n            <jats:italic>Drosophila Sns<\/jats:italic>\n            .\n          <\/jats:p>","DOI":"10.1073\/pnas.0904398106","type":"journal-article","created":{"date-parts":[[2009,5,23]],"date-time":"2009-05-23T01:46:27Z","timestamp":1243043187000},"page":"9274-9279","update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":82,"title":["A role for nephrin, a renal protein, in vertebrate skeletal muscle cell fusion"],"prefix":"10.1073","volume":"106","author":[{"given":"Regina Lee","family":"Sohn","sequence":"first","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"}]},{"given":"Ping","family":"Huang","sequence":"additional","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"}]},{"given":"Genri","family":"Kawahara","sequence":"additional","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"}]},{"given":"Matthew","family":"Mitchell","sequence":"additional","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"}]},{"given":"Jeffrey","family":"Guyon","sequence":"additional","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"}]},{"given":"Raghu","family":"Kalluri","sequence":"additional","affiliation":[{"name":"Division of Matrix Biology, Beth Israel Deaconess Hospital and Department of Biological Chemistry and Molecular Pharmacology, Harvard Medical School and Harvard-MIT Division of Health Sciences and Technology, Boston, MA 02115"}]},{"given":"Louis M.","family":"Kunkel","sequence":"additional","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"},{"name":"The Manton Center for Orphan Disease Research, Children's Hospital Boston and the Howard Hughes Medical Institute, Boston, MA 02115; and"}]},{"given":"Emanuela","family":"Gussoni","sequence":"additional","affiliation":[{"name":"Division of Genetics and Program in Genomics, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115;"}]}],"member":"341","published-online":{"date-parts":[[2009,6,9]]},"reference":[{"key":"e_1_3_3_1_2","doi-asserted-by":"publisher","DOI":"10.1159\/000075028"},{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0854.2008.00756.x"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0074-7696(05)25002-7"},{"key":"e_1_3_3_4_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.1104799"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.1101\/gad.14.12.1498"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.mod.2004.08.001"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1038\/ng0607-704"},{"key":"e_1_3_3_8_2","doi-asserted-by":"publisher","DOI":"10.1038\/ng2055"},{"key":"e_1_3_3_9_2","first-page":"221","article-title":"Nephrin: Role in normal kidney and in disease","volume":"31","author":"Tryggvason K","year":"2001","unstructured":"K Tryggvason, Nephrin: Role in normal kidney and in disease. 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