000830257 001__ 830257 000830257 005__ 20210129230448.0 000830257 0247_ $$2doi$$a10.1074/jbc.M116.773903 000830257 0247_ $$2WOS$$aWOS:000399813400005 000830257 0247_ $$2altmetric$$aaltmetric:19583825 000830257 0247_ $$2pmid$$apmid:28249940 000830257 037__ $$aFZJ-2017-03830 000830257 082__ $$a570 000830257 1001_ $$0P:(DE-HGF)0$$aYerabham, ASK$$b0 000830257 245__ $$aA structural organization for the Disrupted in Schizophrenia 1 protein, identified by high-throughput screening, reveals distinctly folded regions, which are bisected by mental illness-related mutations 000830257 260__ $$aBethesda, Md.$$bSoc.$$c2017 000830257 3367_ $$2DRIVER$$aarticle 000830257 3367_ $$2DataCite$$aOutput Types/Journal article 000830257 3367_ $$0PUB:(DE-HGF)16$$2PUB:(DE-HGF)$$aJournal Article$$bjournal$$mjournal$$s1519723406_17499 000830257 3367_ $$2BibTeX$$aARTICLE 000830257 3367_ $$2ORCID$$aJOURNAL_ARTICLE 000830257 3367_ $$00$$2EndNote$$aJournal Article 000830257 520__ $$aDisrupted in Schizophrenia 1 (DISC1) is a scaffolding protein of significant importance for neurodevelopment and a prominent candidate protein in the pathology of major mental illness. DISC1 modulates a number of critical neuronal signaling pathways through protein-protein interactions; however, the mechanism by which this occurs and how DISC1 causes mental illness is unclear, partly because knowledge of the structure of DISC1 is lacking. A lack of homology with known proteins has hindered attempts to define its domain composition. Here, we employed the high-throughput Expression of Soluble Proteins by Random Incremental Truncation (ESPRIT) technique to identify discretely folded regions of human DISC1 via solubility assessment of tens of thousands of fragments of recombinant DISC1. We identified four novel structured regions, named D, I, S, and C, at amino acids 257–383, 539–655, 635–738, and 691–836, respectively. One region (D) is located in a DISC1 section previously predicted to be unstructured. All regions encompass coiled-coil or α-helical structures, and three are involved in DISC1 oligomerization. Crucially, three of these domains would be lost or disrupted by a chromosomal translocation event after amino acid 597, which has been strongly linked to major mental illness. Furthermore, we observed that a known illness-related frameshift mutation after amino acid 807 causes the C region to form aberrantly multimeric and aggregated complexes with an unstable secondary structure. This newly revealed domain architecture of DISC1, therefore, provides a powerful framework for understanding the critical role of this protein in a variety of devastating mental illnesses. 000830257 536__ $$0G:(DE-HGF)POF3-553$$a553 - Physical Basis of Diseases (POF3-553)$$cPOF3-553$$fPOF III$$x0 000830257 7001_ $$0P:(DE-HGF)0$$aMas, PJ$$b1 000830257 7001_ $$0P:(DE-Juel1)162438$$aDecker, Christina$$b2 000830257 7001_ $$0P:(DE-HGF)0$$aSoares, DC$$b3 000830257 7001_ $$0P:(DE-Juel1)131988$$aWeiergräber, Oliver H.$$b4$$ufzj 000830257 7001_ $$0P:(DE-Juel1)162443$$aNagel-Steger, Luitgard$$b5$$ufzj 000830257 7001_ $$0P:(DE-Juel1)132029$$aWillbold, Dieter$$b6$$ufzj 000830257 7001_ $$0P:(DE-HGF)0$$aHart, DJ$$b7 000830257 7001_ $$0P:(DE-HGF)0$$aBradshaw, NJ$$b8$$eCorresponding author 000830257 7001_ $$0P:(DE-HGF)0$$aKorth, C.$$b9$$eCorresponding author 000830257 773__ $$0PERI:(DE-600)1474604-9$$a10.1074/jbc.M116.773903$$p6468–6477$$tThe journal of biological chemistry$$v292$$x0021-9258$$y2017 000830257 8564_ $$uhttps://juser.fz-juelich.de/record/830257/files/A%20structural%20organization%20for%20the%20Disrupted%20in%20Schizophrenia%201%20protein%2C%20identified%20by%20high-throughput%20screening%2C%20reveals%20distinctly%20folded%20regions%2C%20which%20are%20bisected%20by%20mental%20illness-related%20mutations.pdf$$yRestricted 000830257 8564_ $$uhttps://juser.fz-juelich.de/record/830257/files/A%20structural%20organization%20for%20the%20Disrupted%20in%20Schizophrenia%201%20protein%2C%20identified%20by%20high-throughput%20screening%2C%20reveals%20distinctly%20folded%20regions%2C%20which%20are%20bisected%20by%20mental%20illness-related%20mutations.gif?subformat=icon$$xicon$$yRestricted 000830257 8564_ $$uhttps://juser.fz-juelich.de/record/830257/files/A%20structural%20organization%20for%20the%20Disrupted%20in%20Schizophrenia%201%20protein%2C%20identified%20by%20high-throughput%20screening%2C%20reveals%20distinctly%20folded%20regions%2C%20which%20are%20bisected%20by%20mental%20illness-related%20mutations.jpg?subformat=icon-1440$$xicon-1440$$yRestricted 000830257 8564_ $$uhttps://juser.fz-juelich.de/record/830257/files/A%20structural%20organization%20for%20the%20Disrupted%20in%20Schizophrenia%201%20protein%2C%20identified%20by%20high-throughput%20screening%2C%20reveals%20distinctly%20folded%20regions%2C%20which%20are%20bisected%20by%20mental%20illness-related%20mutations.jpg?subformat=icon-180$$xicon-180$$yRestricted 000830257 8564_ $$uhttps://juser.fz-juelich.de/record/830257/files/A%20structural%20organization%20for%20the%20Disrupted%20in%20Schizophrenia%201%20protein%2C%20identified%20by%20high-throughput%20screening%2C%20reveals%20distinctly%20folded%20regions%2C%20which%20are%20bisected%20by%20mental%20illness-related%20mutations.jpg?subformat=icon-640$$xicon-640$$yRestricted 000830257 8564_ $$uhttps://juser.fz-juelich.de/record/830257/files/A%20structural%20organization%20for%20the%20Disrupted%20in%20Schizophrenia%201%20protein%2C%20identified%20by%20high-throughput%20screening%2C%20reveals%20distinctly%20folded%20regions%2C%20which%20are%20bisected%20by%20mental%20illness-related%20mutations.pdf?subformat=pdfa$$xpdfa$$yRestricted 000830257 909CO $$ooai:juser.fz-juelich.de:830257$$pVDB 000830257 9101_ $$0I:(DE-588b)5008462-8$$6P:(DE-Juel1)131988$$aForschungszentrum Jülich$$b4$$kFZJ 000830257 9101_ $$0I:(DE-588b)5008462-8$$6P:(DE-Juel1)162443$$aForschungszentrum Jülich$$b5$$kFZJ 000830257 9101_ $$0I:(DE-588b)5008462-8$$6P:(DE-Juel1)132029$$aForschungszentrum Jülich$$b6$$kFZJ 000830257 9131_ $$0G:(DE-HGF)POF3-553$$1G:(DE-HGF)POF3-550$$2G:(DE-HGF)POF3-500$$3G:(DE-HGF)POF3$$4G:(DE-HGF)POF$$aDE-HGF$$bKey Technologies$$lBioSoft – Fundamentals for future Technologies in the fields of Soft Matter and Life Sciences$$vPhysical Basis of Diseases$$x0 000830257 9141_ $$y2017 000830257 915__ $$0StatID:(DE-HGF)0200$$2StatID$$aDBCoverage$$bSCOPUS 000830257 915__ $$0StatID:(DE-HGF)0300$$2StatID$$aDBCoverage$$bMedline 000830257 915__ $$0StatID:(DE-HGF)0310$$2StatID$$aDBCoverage$$bNCBI Molecular Biology Database 000830257 915__ $$0StatID:(DE-HGF)0100$$2StatID$$aJCR$$bJ BIOL CHEM : 2015 000830257 915__ $$0StatID:(DE-HGF)0600$$2StatID$$aDBCoverage$$bEbsco Academic Search 000830257 915__ $$0StatID:(DE-HGF)0030$$2StatID$$aPeer Review$$bASC 000830257 915__ $$0StatID:(DE-HGF)0199$$2StatID$$aDBCoverage$$bThomson Reuters Master Journal List 000830257 915__ $$0StatID:(DE-HGF)0110$$2StatID$$aWoS$$bScience Citation Index 000830257 915__ $$0StatID:(DE-HGF)0150$$2StatID$$aDBCoverage$$bWeb of Science Core Collection 000830257 915__ $$0StatID:(DE-HGF)0111$$2StatID$$aWoS$$bScience Citation Index Expanded 000830257 915__ $$0StatID:(DE-HGF)1030$$2StatID$$aDBCoverage$$bCurrent Contents - 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