{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,25]],"date-time":"2026-07-25T11:01:30Z","timestamp":1784977290655,"version":"3.55.0"},"reference-count":78,"publisher":"Wiley","issue":"23","license":[{"start":{"date-parts":[[2013,10,8]],"date-time":"2013-10-08T00:00:00Z","timestamp":1381190400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["febs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The FEBS Journal"],"published-print":{"date-parts":[[2013,12]]},"abstract":"<jats:p>Mammalian muscle cell differentiation is a complex process of multiple steps for which many of the factors involved have not yet been defined. In a screen to identify the regulators of myogenic cell fusion, we found that the gene for <jats:styled-content style=\"fixed-case\">G<\/jats:styled-content>\u2010protein coupled receptor 56 (<jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56) was transiently up\u2010regulated during the early fusion of human myoblasts. Human mutations in the gene for <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56 cause the disease bilateral frontoparietal polymicrogyria; however, the consequences of receptor dysfunction on muscle development have not been explored. Using knockout mice, we defined the role of <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56 in skeletal muscle. <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56<jats:sup>\u2212\/\u2212<\/jats:sup> myoblasts have decreased fusion and smaller myotube sizes in culture. In addition, a loss of <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56 expression in muscle cells results in decreases or delays in the expression of myogenic differentiation 1, myogenin and nuclear factor of activated <jats:styled-content style=\"fixed-case\">T<\/jats:styled-content>\u2010cell (<jats:styled-content style=\"fixed-case\">NFAT<\/jats:styled-content>)c2. Our data suggest that these abnormalities result from decreased <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56\u2010mediated serum response element and <jats:styled-content style=\"fixed-case\">NFAT<\/jats:styled-content> signalling. Despite these changes, no overt differences in phenotype were identified in the muscle of <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56 knockout mice, which presented only a mild but statistically significant elevation of serum creatine kinase compared to wild\u2010type. In agreement with these findings, clinical data from 13 bilateral frontoparietal polymicrogyria patients revealed mild serum creatine kinase increase in only two patients. In summary, targeted disruption of <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56 in mice results in myoblast abnormalities. The absence of a severe muscle phenotype in <jats:styled-content style=\"fixed-case\">GPR<\/jats:styled-content>56 knockout mice and human patients suggests that other factors may compensate for the lack of this <jats:styled-content style=\"fixed-case\">G<\/jats:styled-content>\u2010protein coupled receptor during muscle development and that the motor delay observed in these patients is likely not a result of primary muscle abnormalities.<\/jats:p>","DOI":"10.1111\/febs.12529","type":"journal-article","created":{"date-parts":[[2013,10,8]],"date-time":"2013-10-08T15:25:30Z","timestamp":1381245930000},"page":"6097-6113","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":44,"title":["G\u2010protein coupled receptor 56 promotes myoblast fusion through serum response factor\u2010 and nuclear factor of activated T\u2010cell\u2010mediated signalling but is not essential for muscle development <i>in\u00a0vivo<\/i>"],"prefix":"10.1111","volume":"280","author":[{"given":"Melissa P.","family":"Wu","sequence":"first","affiliation":[{"name":"Department of Biological and Biomedical Sciences Harvard Medical School Boston MA USA"},{"name":"Division of Genetics Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jamie R.","family":"Doyle","sequence":"additional","affiliation":[{"name":"Molecular Cardiology Research Institute Tufts Medical Center Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brenda","family":"Barry","sequence":"additional","affiliation":[{"name":"Division of Genetics Boston Children's Hospital MA USA"},{"name":"Howard Hughes Medical Institute Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ariane","family":"Beauvais","sequence":"additional","affiliation":[{"name":"Division of Genetics Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anete","family":"Rozkalne","sequence":"additional","affiliation":[{"name":"Division of Genetics Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xianhua","family":"Piao","sequence":"additional","affiliation":[{"name":"Division of Newborn Medicine Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael W.","family":"Lawlor","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine Children's Hospital of Wisconsin and Medical College of Wisconsin Milwaukee WI USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alan S.","family":"Kopin","sequence":"additional","affiliation":[{"name":"Molecular Cardiology Research Institute Tufts Medical Center Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher A.","family":"Walsh","sequence":"additional","affiliation":[{"name":"Division of Genetics Boston Children's Hospital MA USA"},{"name":"Howard Hughes Medical Institute Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emanuela","family":"Gussoni","sequence":"additional","affiliation":[{"name":"Division of Genetics Boston Children's Hospital MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2013,10,8]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1469-7580.2003.00139.x"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.068353"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/0955-0674(94)90046-9"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.111.4.1097"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.7863327"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/j.1460-2075.1996.tb00361.x"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.124.23.4729"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(92)90508-A"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(93)90621-V"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(89)90583-7"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yexcr.2010.05.008"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.03056"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E03-12-0886"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.146.4.893"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.onc.1208395"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1158\/1535-7163.MCT-07-0066"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M708919200"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.0853-08.2008"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1182-09.2009"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10585-010-9322-3"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1104821108"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-4419-7913-1_4"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1038\/emboj.2012.26"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0896-6273(00)80332-3"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M004814200"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M111.247973"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1092780"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddm144"},{"key":"e_1_2_7_30_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-2008-1071501"},{"key":"e_1_2_7_31_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-2007-973752"},{"key":"e_1_2_7_32_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1996.tb02721.x"},{"key":"e_1_2_7_33_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1997.tb02482.x"},{"key":"e_1_2_7_34_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.20616"},{"key":"e_1_2_7_35_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awp247"},{"key":"e_1_2_7_36_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10048-012-0352-7"},{"key":"e_1_2_7_37_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature00837"},{"key":"e_1_2_7_38_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000115386.28769.65"},{"key":"e_1_2_7_39_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1750-3639.2008.00198.x"},{"key":"e_1_2_7_40_1","doi-asserted-by":"publisher","DOI":"10.1096\/fj.04-2215fje"},{"key":"e_1_2_7_41_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00432-009-0635-z"},{"key":"e_1_2_7_42_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.106.1.67"},{"key":"e_1_2_7_43_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajpath.2012.05.016"},{"key":"e_1_2_7_44_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.7.5.719"},{"key":"e_1_2_7_45_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E02-07-0451"},{"key":"e_1_2_7_46_1","first-page":"41","article-title":"Identification of new SRF binding sites in genes modulated by SRF over\u2010expression in mouse hearts","volume":"5","author":"Zhang X","year":"2011","journal-title":"Gene Regul Syst Bio"},{"key":"e_1_2_7_47_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.153.2.329"},{"key":"e_1_2_7_48_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00401-002-0647-0"},{"key":"e_1_2_7_49_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200804077"},{"key":"e_1_2_7_50_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awq259"},{"key":"e_1_2_7_51_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.9.7.1891"},{"key":"e_1_2_7_52_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.20.17.6600-6611.2000"},{"key":"e_1_2_7_53_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2001.0179"},{"key":"e_1_2_7_54_1","doi-asserted-by":"publisher","DOI":"10.1152\/ajpcell.00386.2006"},{"key":"e_1_2_7_55_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0308035101"},{"key":"e_1_2_7_56_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0812911106"},{"key":"e_1_2_7_57_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1528-1167.2008.01787.x"},{"key":"e_1_2_7_58_1","doi-asserted-by":"publisher","DOI":"10.1093\/emboj\/18.4.863"},{"key":"e_1_2_7_59_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.febslet.2005.03.033"},{"key":"e_1_2_7_60_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0505338103"},{"key":"e_1_2_7_61_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.118.6.1489"},{"key":"e_1_2_7_62_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.10.10.1173"},{"key":"e_1_2_7_63_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1999.9284"},{"key":"e_1_2_7_64_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.27.3.181"},{"key":"e_1_2_7_65_1","doi-asserted-by":"publisher","DOI":"10.1016\/0009-8981(65)90186-5"},{"key":"e_1_2_7_66_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddq314"},{"key":"e_1_2_7_67_1","doi-asserted-by":"publisher","DOI":"10.1152\/jappl.1994.77.2.493"},{"key":"e_1_2_7_68_1","first-page":"117","article-title":"The generation of fiber diversity during myogenesis","volume":"42","author":"Wigmore PM","year":"1998","journal-title":"Int J Dev Biol"},{"key":"e_1_2_7_69_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.93.2.442"},{"key":"e_1_2_7_70_1","doi-asserted-by":"publisher","DOI":"10.1002\/j.1460-2075.1985.tb03755.x"},{"key":"e_1_2_7_71_1","doi-asserted-by":"publisher","DOI":"10.1002\/cne.903230208"},{"key":"e_1_2_7_72_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.R200027200"},{"key":"e_1_2_7_73_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0012-1606(81)80010-3"},{"key":"e_1_2_7_74_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.21785"},{"key":"e_1_2_7_75_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF02721371"},{"key":"e_1_2_7_76_1","doi-asserted-by":"publisher","DOI":"10.1006\/meth.2001.1262"},{"key":"e_1_2_7_77_1","doi-asserted-by":"publisher","DOI":"10.1038\/nprot.2006.236"},{"key":"e_1_2_7_78_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M806979200"},{"key":"e_1_2_7_79_1","doi-asserted-by":"publisher","DOI":"10.1124\/jpet.112.194548"}],"container-title":["The FEBS Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Ffebs.12529","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/febs.onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/febs.12529","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,3]],"date-time":"2023-10-03T05:57:06Z","timestamp":1696312626000},"score":1,"resource":{"primary":{"URL":"https:\/\/febs.onlinelibrary.wiley.com\/doi\/10.1111\/febs.12529"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,10,8]]},"references-count":78,"journal-issue":{"issue":"23","published-print":{"date-parts":[[2013,12]]}},"alternative-id":["10.1111\/febs.12529"],"URL":"https:\/\/doi.org\/10.1111\/febs.12529","archive":["Portico"],"relation":{},"ISSN":["1742-464X","1742-4658"],"issn-type":[{"value":"1742-464X","type":"print"},{"value":"1742-4658","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,10,8]]},"assertion":[{"value":"2013-07-01","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2013-09-04","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2013-10-08","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}