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The approach usually involves expressing the mutant form of the gene in cells and assaying function in a multiwell format when cells are exposed to libraries of compounds. Although such functional assays are useful, they do not directly test the ability of a compound to correct defective trafficking of the protein. To address this we have developed a novel corrector\u2010screening assay for CF, in which the appearance of the mutant protein at the cell surface is measured. We used this assay to screen a library of 2000 compounds and have isolated several classes of trafficking correctors that had not previously been identified. This novel screening approach to protein\u2010trafficking diseases is robust and general, and could enable the selection of molecules that could be translated rapidly to a clinical setting.<\/jats:p>","DOI":"10.1002\/cbic.200700027","type":"journal-article","created":{"date-parts":[[2007,5,11]],"date-time":"2007-05-11T08:57:26Z","timestamp":1178873846000},"page":"1012-1020","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":93,"title":["Correctors of Protein Trafficking Defects Identified by a Novel High\u2010Throughput Screening Assay"],"prefix":"10.1002","volume":"8","author":[{"given":"Graeme W.","family":"Carlile","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Renaud","family":"Robert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donglei","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katrina A.","family":"Teske","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yishan","family":"Luo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John W.","family":"Hanrahan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David Y.","family":"Thomas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2007,6]]},"reference":[{"key":"e_1_2_7_1_2","unstructured":"\u00a0"},{"key":"e_1_2_7_2_2","first-page":"1867","volume-title":"The Metabolic Basis of Inherited Disease","author":"Insipidus\u201d \u201cNephrogenic Diabetes","year":"1983"},{"key":"e_1_2_7_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(95)90340-2"},{"key":"e_1_2_7_4_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.2475911"},{"key":"e_1_2_7_5_2","unstructured":"\u00a0"},{"key":"e_1_2_7_6_2","doi-asserted-by":"publisher","DOI":"10.1172\/JCI24898"},{"key":"e_1_2_7_7_2","doi-asserted-by":"publisher","DOI":"10.1152\/ajpcell.2001.281.5.C1734"},{"key":"e_1_2_7_8_2","unstructured":"Report of the Canadian Cystic Fibrosis Patient Data Registry C.C.F.F.2002."},{"key":"e_1_2_7_9_2","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(92)90155-6"},{"key":"e_1_2_7_10_2","unstructured":"CFTR mutation database:http:\/\/www.genet.sickkids.on.ca\/cftr\/."},{"key":"e_1_2_7_11_2","unstructured":"\u00a0"},{"key":"e_1_2_7_12_2","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.118.3.551"},{"key":"e_1_2_7_13_2","doi-asserted-by":"publisher","DOI":"10.1379\/1466-1268(1996)001<0117:CCCTMP>2.3.CO;2"},{"key":"e_1_2_7_14_2","doi-asserted-by":"publisher","DOI":"10.1172\/JCI119788"},{"key":"e_1_2_7_15_2","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M508944200"},{"key":"e_1_2_7_16_2","doi-asserted-by":"publisher","DOI":"10.1152\/ajplung.00049.2005"},{"key":"e_1_2_7_17_2","doi-asserted-by":"publisher","DOI":"10.1152\/ajpcell.1995.269.6.C1565"},{"key":"e_1_2_7_18_2","doi-asserted-by":"publisher","DOI":"10.1165\/ajrcmb\/8.5.522"},{"key":"e_1_2_7_19_2","doi-asserted-by":"publisher","DOI":"10.1006\/mthe.2000.0095"},{"key":"e_1_2_7_20_2","doi-asserted-by":"publisher","DOI":"10.1113\/jphysiol.2005.096669"},{"key":"e_1_2_7_21_2","doi-asserted-by":"publisher","DOI":"10.1152\/ajplung.2001.281.1.L24"},{"key":"e_1_2_7_22_2","doi-asserted-by":"publisher","DOI":"10.1165\/rcmb.2002-0247OC"},{"key":"e_1_2_7_23_2","doi-asserted-by":"publisher","DOI":"10.1007\/s004240050909"},{"key":"e_1_2_7_24_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1981.tb05731.x"},{"key":"e_1_2_7_25_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2004.02.002"},{"key":"e_1_2_7_26_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbrc.2006.03.009"},{"key":"e_1_2_7_27_2","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1007\/BF03402077","volume":"8","author":"Carvalho A. 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