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PageLayout: SinglePage PageMode: UseOutlines Language: EN Outlines: Item: Title: The human ciliopathy protein RSG1 links the CPLANE complex to transition zone architecture Destination: title_link Children: Item: Title: Results Destination: A1 Children: Item: Title: Allelic variants of CPLANE2/RSG1 cause human ciliopathy Destination: B1 Item: Title: RSG1 variants disrupt RSG1 function via distinct mechanisms Destination: B2 Item: Title: RSG1 variants disrupt IFT-A2 recruitment to basal bodies Destination: B3 Item: Title: RSG1 and JBTS17 variants disrupt basal body docking Destination: B4 Item: Title: GTP binding is required for Rsg1 association with CPLANE Destination: B5 Item: Title: The GTP-dependent Rsg1 interactome Destination: B6 Item: Title: AlphaFold predicts direct RSG1/FAM92A interaction Destination: B7 Item: Title: Rsg1 recruits Fam92b specifically to docking basal bodies Destination: B8 Item: Title: Human RSG1 controls ciliogenesis and FAM92a recruitment Destination: B9 Item: Title: CPLANE proteins recruit transition zone components Destination: B10 Item: Title: Discussion Destination: A2 Item: Title: Methods Destination: A3 Children: Item: Title: Patients Destination: B11 Item: Title: Patient genetic analysis Destination: B12 Item: Title: Xenopus embryo manipulations Destination: B13 Item: Title: Plasmid, mRNA, and MO microinjections Destination: B14 Item: Title: In vitro pull-down assay Destination: B15 Item: Title: Immunoblotting Destination: B16 Item: Title: Live imaging and image analysis for Xenopus Destination: B17 Item: Title: Tandem affinity purification Destination: B18 Item: Title: Mass spectrometry Destination: B19 Item: Title: Data analysis Destination: B20 Item: Title: Protein structure modeling Destination: B21 Item: Title: Human and mouse cell culture Destination: B22 Item: Title: Immunostaining Destination: B23 Item: Title: Antibodies Destination: B24 Item: Title: Image analysis of NPHP1 and ciliogenesis Destination: B25 Item: Title: Reporting summary Destination: B26 Item: Title: Data availability Destination: M1 Item: Title: References Destination: A4 Item: Title: Acknowledgements Destination: A5 Item: Title: Author contributions Destination: A6 Item: Title: Competing interests Destination: A7 Item: Title: Additional information Destination: M3 Info: Title: The human ciliopathy protein RSG1 links the CPLANE complex to transition zone architecture Author: Neftalí Vazquez Subject: Nature Communications, doi:10.1038/s41467-025-61005-8 Keywords: Creator: Springer Producer: iText® 5.5.13.4 ©2000-2024 iText Group NV (SPRINGER SBM; licensed version) CreationDate: Wed Jan 28 05:11:19 CET 2026 ModDate: Tue Feb 03 07:37:59 CET 2026 ID: 0xa506d0dc757cdc2462ad7c462be509ea, 0x0c4439f4807374beb2cf3e1086a7ab0e Filters: FilterPipeline: FlateDecode FilterPipeline: DCTDecode Images: Image: NisoImageMetadata: FormatName: image/png ImageWidth: 46 ImageHeight: 40 BitsPerSample: 8 BitsPerSampleUnit: integer Filter: FlateDecode Image: 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Phan Camille Derderian Marcelo Chávez Nancie A. Mooney Janos Demeter Mohammad Ovais Aziz-Zanjani Ivon Cusco Marta Codina Núria Martínez-Gil Diana Valverde Carlos Solarat Ange-Line Bruel Cristel Thauvin-Robinet Elisabeth Steichen Isabel Filges Pascal Joset Julie Geyter Krishna Vaidyanathan Tynan P. Gardner Michinori Toriyama Edward M. Marcotte Kevin Drew Elle C. Roberson Peter K. Jackson Jeremy F. Reiter Eduardo F. Tizzano John B. 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