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A major biological effect of<jats:italic>O<\/jats:italic>\u2010mannosylation involves the regulation of \u03b1\u2010dystroglycan, a membrane glycoprotein mediating cell\u2013extracellular matrix interactions. Genetic defects of<jats:italic>O<\/jats:italic>\u2010mannosylation result in the loss of ligand\u2010binding activity of \u03b1\u2010dystroglycan and cause congenital muscular dystrophies termed dystroglycanopathies. Recent progress in mass spectrometry and in vitro analyses has shed new light on the mechanism of \u03b1\u2010dystroglycan glycosylation; however, this mechanism is underlain by complex genetic and molecular elements that remain poorly understood. Protein<jats:italic>O<\/jats:italic>\u2010mannosylation is evolutionarily conserved in metazoans, yet the pathway is simplified and more amenable to genetic analyses in invertebrate organisms, indicating that genetically tractable in vivo models could facilitate research in this area. This unit describes recent methodological strategies for studying protein<jats:italic>O<\/jats:italic>\u2010mannosylation using in vitro and in vivo approaches.<jats:italic>Curr. Protoc. Protein Sci<\/jats:italic>. 75:12.12.1\u201012.12.29. \u00a9 2014 by John Wiley &amp; Sons, Inc.<\/jats:p>","DOI":"10.1002\/0471140864.ps1212s75","type":"journal-article","created":{"date-parts":[[2014,2,3]],"date-time":"2014-02-03T14:33:07Z","timestamp":1391437987000},"source":"Crossref","is-referenced-by-count":12,"title":["Protein<i>O<\/i>\u2010Mannosylation in Metazoan Organisms"],"prefix":"10.1002","volume":"75","author":[{"given":"Vladislav M.","family":"Panin","sequence":"first","affiliation":[{"name":"Department of Biochemistry and Biophysics, Texas A&amp;M University College Station Texas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lance","family":"Wells","sequence":"additional","affiliation":[{"name":"Complex Carbohydrate Research Center, Department of Biochemistry and Molecular Biology, University of Georgia Athens Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,2,3]]},"reference":[{"key":"e_1_2_17_2_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M803646200"},{"key":"e_1_2_17_3_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M601091200"},{"key":"e_1_2_17_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/0003-2697(92)90048-C"},{"key":"e_1_2_17_5_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M804925200"},{"key":"e_1_2_17_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.23632"},{"key":"e_1_2_17_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/nm1059"},{"key":"e_1_2_17_8_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI63004"},{"key":"e_1_2_17_9_1","doi-asserted-by":"publisher","DOI":"10.1086\/342975"},{"key":"e_1_2_17_10_1","doi-asserted-by":"publisher","DOI":"10.1515\/BC.2009.044"},{"key":"e_1_2_17_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0091-679X(08)60936-X"},{"key":"e_1_2_17_12_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M802834200"},{"key":"e_1_2_17_13_1","doi-asserted-by":"publisher","DOI":"10.1086\/324412"},{"key":"e_1_2_17_14_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/10.25.2851"},{"key":"e_1_2_17_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddi062"},{"key":"e_1_2_17_16_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddt021"},{"key":"e_1_2_17_17_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.272.4.2156"},{"key":"e_1_2_17_18_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.C900007200"},{"key":"e_1_2_17_19_1","doi-asserted-by":"publisher","DOI":"10.1093\/genetics\/31.3.269"},{"key":"e_1_2_17_20_1","first-page":"43","article-title":"O\u2010Mannosylation in mammalian cells","volume":"347","author":"Endo T.","year":"2006","journal-title":"Methods Mol. 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