WDR31 Displays Functional Redundancy With GTpase-Activating Proteins (GAPs) ELMOD and RP2 in Regulating Ift Complex and Recruiting the BBsome to Cilium

dc.contributor.author Cevik, Sebiha
dc.contributor.author Peng, Xiaoyu
dc.contributor.author Beyer, Tina
dc.contributor.author Pir, Mustafa S.
dc.contributor.author Yenisert, Ferhan
dc.contributor.author Woerz, Franziska
dc.contributor.author Kaplan, Oktay, I
dc.date.accessioned 2025-09-25T11:00:56Z
dc.date.available 2025-09-25T11:00:56Z
dc.date.issued 2023
dc.description Beyer, Tina/0000-0002-6302-8997; Cakiroglu, Miray/0000-0002-4289-8344; Oner, Sukru Sadik/0000-0002-8864-7356; Woerz, Franziska/0009-0007-5484-358X; Kaplan, Oktay Ismail/0000-0002-8733-0920; Hoffmann, Felix/0000-0001-5199-2749; Yenisert, Ferhan/0000-0002-1028-8197; Pir, Mustafa Samet/0000-0002-4645-7626; Cevik, Sebiha/0000-0002-0935-1929 en_US
dc.description.abstract The correct intraflagellar transport (IFT) assembly at the ciliary base and the IFT turnaround at the ciliary tip are key for the IFT to perform its function, but we still have poor understanding about how these processes are regulated. Here, we identify WDR31 as a new ciliary protein, and analysis from zebrafish and Caeno-rhabditis elegans reveals the role of WDR31 in regulating the cilia morphology. We find that loss of WDR-31 together with RP-2 and ELMD-1 (the sole ortholog ELMOD1-3) results in ciliary accumu-lations of IFT Complex B components and KIF17 kinesin, with fewer IFT/BBSome particles traveling along cilia in both anterograde and retrograde directions, suggesting that the IFT/BBSome entry into the cilia and exit from the cilia are impacted. Furthermore, anterograde IFT in the middle segment travels at increased speed in wdr-31;rpi-2;elmd-1. Remarkably, a non-ciliary protein leaks into the cilia of wdr-31;rpi-2;elmd-1, possibly because of IFT de-fects. This work reveals WDR31-RP-2-ELMD-1 as IFT and BBSome trafficking regulators. en_US
dc.description.sponsorship National BioResource Project (NBRP) in Japan; NIH Of fice of Research Infrastructure Programs [P40 OD010440]; Abdullah Gul University Scienti fic Research Project Coordination Unit [TOA-2018-110]; Tistou AMP; Charlotte Kerstan Stiftung; Deutsche Forschungsgemeinschaft; [INST 2388/62-1] en_US
dc.description.sponsorship We thank Oliver Blacque, Piali Sengupta, Micheal Leroux, the National BioResource Project (NBRP) in Japan, and the CGC in the United States, which is financed by the NIH Of fice of Research Infrastructure Programs (P40 OD010440) for sharing valuable strains. We thank Atiyye Zorluer for gen- erating strains. We thank Oliver Blacque and Samuel Katz for critical reading of the manuscript. We thank the Abdullah Gul University Scienti fic Research Project Coordination Unit (Project number: TOA-2018-110) for providing the funding that initiated the project. M Uef fing was supported by the Tistou & Charlotte Kerstan Stiftung. The Leica laser scanning microscope was funded by a grant from Deutsche Forschungsgemeinschaft (INST 2388/62-1) . en_US
dc.description.sponsorship National BioResource Project (NBRP) in Japan; NIH Of fice of Research Infrastructure Programs [P40 OD010440]; Abdullah Gul University Scienti fic Research Project Coordination Unit [TOA-2018-110]; Tistou AMP; Charlotte Kerstan Stiftung; Deutsche Forschungsgemeinschaft; [INST 2388/62-1]; National Institute of General Medical Sciences; NIH Office of the Director [P40OD010440] Funding Source: NIH RePORTER
dc.description.sponsorship We thank Oliver Blacque, Piali Sengupta, Micheal Leroux, the National BioResource Project (NBRP) in Japan, and the CGC in the United States, which is financed by the NIH Office of Research Infrastructure Programs (P40 OD010440) for sharing valuable strains. We thank Atiyye Zorluer for generating strains. We thank Oliver Blacque and Samuel Katz for critical reading of the manuscript. We thank the Abdullah Gul University Scientific Research Project Coordination Unit (Project number: TOA-2018-110) for providing the funding that initiated the project. M Ueffing was supported by the Tistou & Charlotte Kerstan Stiftung. The Leica laser scanning microscope was funded by a grant from Deutsche Forschungsgemeinschaft (INST 2388/62-1).
dc.description.sponsorship Abdullah Gul University, (TOA-2018-110); National Institutes of Health, NIH, (P40 OD010440); Deutsche Forschungsgemeinschaft, DFG, (INST 2388/62-1); Tistou and Charlotte Kerstan Stiftung
dc.identifier.doi 10.26508/lsa.202201844
dc.identifier.issn 2575-1077
dc.identifier.scopus 2-s2.0-85159654316
dc.identifier.uri https://doi.org/10.26508/lsa.202201844
dc.identifier.uri https://hdl.handle.net/20.500.12573/4956
dc.language.iso en en_US
dc.publisher Life Science Alliance Llc en_US
dc.relation.ispartof Life Science Alliance en_US
dc.rights info:eu-repo/semantics/openAccess en_US
dc.title WDR31 Displays Functional Redundancy With GTpase-Activating Proteins (GAPs) ELMOD and RP2 in Regulating Ift Complex and Recruiting the BBsome to Cilium en_US
dc.type Article en_US
dspace.entity.type Publication
gdc.author.id Beyer, Tina/0000-0002-6302-8997
gdc.author.id Cakiroglu, Miray/0000-0002-4289-8344
gdc.author.id Oner, Sukru Sadik/0000-0002-8864-7356
gdc.author.id Woerz, Franziska/0009-0007-5484-358X
gdc.author.id Kaplan, Oktay Ismail/0000-0002-8733-0920
gdc.author.id Hoffmann, Felix/0000-0001-5199-2749
gdc.author.id Cevik, Sebiha/0000-0002-0935-1929
gdc.author.id PİR, MUSTAFA SAMET/0000-0002-4645-7626
gdc.author.id YENİSERT, FERHAN/0000-0002-1028-8197
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gdc.author.scopusid 23472963100
gdc.author.wosid Oner, Sukru/K-8032-2012
gdc.author.wosid Peng, Xiaoyu/Iun-3010-2023
gdc.author.wosid Beyer, Tina/Aag-1202-2021
gdc.author.wosid Kaplan, Oktay Ismail/F-8531-2015
gdc.author.wosid Pir, Mustafa/Aeo-1681-2022
gdc.author.wosid Kaplan, Oktay/F-8531-2015
gdc.author.wosid YENİSERT, FERHAN/PMR-2493-2026
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gdc.coar.access open access
gdc.coar.type text::journal::journal article
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gdc.description.department Abdullah Gül University en_US
gdc.description.departmenttemp [Cevik, Sebiha; Pir, Mustafa S.; Yenisert, Ferhan; Altunkaynak, Betul; Pir, Betul; Kaplan, Oktay, I] Abdullah Gul Univ, Sch Life & Nat Sci, Rare Dis Lab, Kayseri, Turkiye; [Peng, Xiaoyu; Cao, Ying] Tongji Univ, Sch Life Sci & Technol, Shanghai, Peoples R China; [Beyer, Tina; Woerz, Franziska; Hoffmann, Felix] Univ Tubingen, Inst Ophthalm Res, Ctr Ophthalmol, Tubingen, Germany; [Oner, S. Sadik] Goztepe Prof Dr Suleyman Yalcin City Hosp, Istanbul, Turkiye; [Karaman, Asli; Cakiroglu, Miray; Oner, S. Sadik] Istanbul Medeniyet Univ, Sci & Adv Technol Applicat & Res Ctr, Istanbul, Turkiye en_US
gdc.description.issue 8 en_US
gdc.description.publicationcategory Makale - Uluslararası Hakemli Dergi - Kurum Öğretim Elemanı en_US
gdc.description.scopusquality Q2
gdc.description.startpage e202201844
gdc.description.volume 6 en_US
gdc.description.woscitationindex Science Citation Index Expanded
gdc.description.wosquality Q2
gdc.identifier.openalex W4377142795
gdc.identifier.pmid 37208194
gdc.identifier.wos WOS:000995885500002
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gdc.index.type PubMed
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gdc.oaire.diamondjournal false
gdc.oaire.downloads 80
gdc.oaire.impulse 7.0
gdc.oaire.influence 2.6684737E-9
gdc.oaire.isgreen true
gdc.oaire.keywords A COMPLEX
gdc.oaire.keywords RAFFICKING
gdc.oaire.keywords GTPase-Activating Proteins
gdc.oaire.keywords ZONE
gdc.oaire.keywords Biological Transport
gdc.oaire.keywords Zebrafish Proteins
gdc.oaire.keywords CILIARY GENES
gdc.oaire.keywords INTRAFLAGELLAR TRANSPORT
gdc.oaire.keywords PHOSPHOINOSITIDEST
gdc.oaire.keywords Animals
gdc.oaire.keywords Cilia
gdc.oaire.keywords CAENORHABDITIS-ELEGANS
gdc.oaire.keywords Caenorhabditis elegans
gdc.oaire.keywords Caenorhabditis elegans Proteins
gdc.oaire.keywords GENOMICS
gdc.oaire.keywords Research Articles
gdc.oaire.keywords Zebrafish
gdc.oaire.keywords CILIOGENESIS
gdc.oaire.popularity 7.2901813E-9
gdc.oaire.publicfunded false
gdc.oaire.views 132
gdc.openalex.collaboration International
gdc.openalex.fwci 2.5724
gdc.openalex.normalizedpercentile 0.9
gdc.openalex.toppercent TOP 10%
gdc.opencitations.count 6
gdc.plumx.mendeley 5
gdc.plumx.pubmedcites 3
gdc.plumx.scopuscites 5
gdc.scopus.citedcount 5
gdc.virtual.author Çevik Kaplan, Sebiha
gdc.virtual.author Kaplan, Oktay İsmail
gdc.wos.citedcount 5
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