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A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function.
Hong J, Lee C, Madhu G, Papoulas O, Atayeter E, Hoogerbrugge G, Pan J, Takagishi M, Manzi NI, Dickinson DJ, Horani A, Brody SL, Marcotte EM, Prakash VN, Park TJ, Wallingford JB. Hong J, et al. Among authors: wallingford jb. Nat Commun. 2025 Dec 21;17(1):394. doi: 10.1038/s41467-025-67086-9. Nat Commun. 2025. PMID: 41423487 Free PMC article.
The beating of cilia on multi-ciliated cells (MCCs) is essential for normal development and homeostasis in animals. ...Ccdc78 and Ccdc33 each display robust microtubule-bundling activity both in vivo and in vitro, yet each is independently required for normal length regula …
The beating of cilia on multi-ciliated cells (MCCs) is essential for normal development and homeostasis in animals. ...Ccdc78 and Ccd …
Cilia.io: Computer vision and machine learning reveal spatial patterns of cilia beating dynamics in the spinal cord.
Atayeter E, Ho J, Blottin TG, Joe IB, Sistrunk RS, Zhang B, Solnica-Krezel L, Gerstlauer A, Wallingford JB, Gray RS. Atayeter E, et al. Among authors: wallingford jb. bioRxiv [Preprint]. 2025 Dec 13:2025.12.10.693099. doi: 10.64898/2025.12.10.693099. bioRxiv. 2025. PMID: 41415440 Free PMC article. Preprint.
Cilia.io enables fast, accurate, and reproducible quantification of motile cilia morphodynamics and outperforms existing tools. ...In these mutants, only dorsal cilia displayed altered beating dynamics, while ventral cilia remained largely unaffected. …
Cilia.io enables fast, accurate, and reproducible quantification of motile cilia morphodynamics and outperforms existing tools …
Identification of conserved residues essential for the ciliogenic functions of WDPCP.
Choi YJ, Hwang S, Lee C, Zeng H, Chen X, Jo U, Kim H, Liu A, Park D, Wallingford JB, Chen J. Choi YJ, et al. Among authors: wallingford jb. Dis Model Mech. 2025 Nov 1;18(11):dmm052149. doi: 10.1242/dmm.052149. Epub 2025 Nov 21. Dis Model Mech. 2025. PMID: 41268724 Free PMC article.
The mutant WDPCP protein was expressed but failed to dock to the apical surface of the cell. Cilia formation and Hh signaling were severely impaired. Structure predictions located these residues at the juncture of two alpha helices in a conserved, but otherwise uncharacter …
The mutant WDPCP protein was expressed but failed to dock to the apical surface of the cell. Cilia formation and Hh signaling were se …
Physical and functional interaction of the ciliopathy proteins Lrrc56 and Odad3 control deployment of axonemal dyneins in vertebrate multiciliated cells.
Reyes-Nava NG, Lee C, Papoulas O, Hong J, Marcotte EM, Wallingford JB. Reyes-Nava NG, et al. Among authors: wallingford jb. Dis Model Mech. 2025 Dec 1;18(12):dmm052523. doi: 10.1242/dmm.052523. Epub 2025 Dec 19. Dis Model Mech. 2025. PMID: 41229303 Free PMC article.
Primary ciliary dyskinesia (PCD) is a genetically heterogeneous motile ciliopathy characterized by chronic respiratory disease, laterality defects, hydrocephalus and infertility, caused by impaired function of motile cilia. LRRC56 has recently emerged as a novel PCD candid …
Primary ciliary dyskinesia (PCD) is a genetically heterogeneous motile ciliopathy characterized by chronic respiratory disease, laterality d …
DynAPs and cytoplasmic assembly of axonemal dyneins.
Wallingford JB, Brody SL, Horani A, Lee C. Wallingford JB, et al. J Cell Sci. 2025 Oct 15;138(20):jcs264137. doi: 10.1242/jcs.264137. Epub 2025 Oct 24. J Cell Sci. 2025. PMID: 41134057
Motile cilia are microtubule-based organelles that generate fluid flow through coordinated beating, a process powered by axonemal dynein motors. ...
Motile cilia are microtubule-based organelles that generate fluid flow through coordinated beating, a process powered by axonemal dyn …
Physical and functional interaction of Lrrc56 and Odad3 controls deployment of axonemal dyneins in vertebrate multiciliated cells.
Reyes-Nava NG, Lee C, Papoulas O, Hong J, Marcotte EM, Wallingford JB. Reyes-Nava NG, et al. Among authors: wallingford jb. bioRxiv [Preprint]. 2025 Jul 3:2025.07.02.662827. doi: 10.1101/2025.07.02.662827. bioRxiv. 2025. Update in: Dis Model Mech. 2025 Dec 1;18(12):dmm052523. doi: 10.1242/dmm.052523. PMID: 40631331 Free PMC article. Updated. Preprint.
Primary ciliary dyskinesia is a genetically heterogeneous motile ciliopathy characterized chronic respiratory disease, laterality defects, hydrocephalus, and infertility, caused by impaired function of motile cilia. LRRC56 has recently emerged as a novel PCD candidate gene …
Primary ciliary dyskinesia is a genetically heterogeneous motile ciliopathy characterized chronic respiratory disease, laterality defects, h …
The human ciliopathy protein RSG1 links the CPLANE complex to transition zone architecture.
Vazquez N, Lee C, Valenzuela I, Phan TP, Derderian C, Chávez M, Mooney NA, Demeter J, Aziz-Zanjani MO, Cusco I, Codina M, Martínez-Gil N, Valverde D, Solarat C, Bruel AL, Thauvin-Robinet C, Steichen E, Filges I, Joset P, De Geyter J, Vaidyanathan K, Gardner TP, Toriyama M, Marcotte EM, Drew K, Roberson EC, Jackson PK, Reiter JF, Tizzano EF, Wallingford JB. Vazquez N, et al. Among authors: wallingford jb. Nat Commun. 2025 Jul 1;16(1):5701. doi: 10.1038/s41467-025-61005-8. Nat Commun. 2025. PMID: 40593758 Free PMC article.
Cilia are essential organelles, and variants in genes governing ciliary function result in ciliopathic diseases. ...
Cilia are essential organelles, and variants in genes governing ciliary function result in ciliopathic diseases. ...
A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function.
Hong J, Lee C, Madhu G, Papoulas O, Atayeter E, Hoogerbrugge G, Pan J, Takagishi M, Manzi N, Dickinson DJ, Horani A, Brody SL, Marcotte E, Prakash VN, Park TJ, Wallingford JB. Hong J, et al. Among authors: wallingford jb. bioRxiv [Preprint]. 2025 Jun 25:2025.02.19.639145. doi: 10.1101/2025.02.19.639145. bioRxiv. 2025. Update in: Nat Commun. 2025 Dec 21;17(1):394. doi: 10.1038/s41467-025-67086-9. PMID: 40027778 Free PMC article. Updated. Preprint.
The beating of cilia on multi-ciliated cells (MCCs) is essential for normal development and homeostasis in animals. ...Ccdc78 and Ccdc33 each display robust microtubule-bundling activity both in vivo and in vitro, yet each is independently required for normal length regula …
The beating of cilia on multi-ciliated cells (MCCs) is essential for normal development and homeostasis in animals. ...Ccdc78 and Ccd …
An amino acid-resolution interactome for motile cilia identifies the structure and function of ciliopathy protein complexes.
McCafferty CL, Papoulas O, Lee C, Bui KH, Taylor DW, Marcotte EM, Wallingford JB. McCafferty CL, et al. Among authors: wallingford jb. Dev Cell. 2025 Mar 24;60(6):965-978.e3. doi: 10.1016/j.devcel.2024.11.019. Epub 2024 Dec 13. Dev Cell. 2025. PMID: 39674175
Motile cilia are ancient, evolutionarily conserved organelles whose dysfunction underlies motile ciliopathies, a broad class of human diseases. Motile cilia contain a myriad of different proteins that assemble into an array of distinct machines, and understanding th …
Motile cilia are ancient, evolutionarily conserved organelles whose dysfunction underlies motile ciliopathies, a broad class of human …
The human ciliopathy protein RSG1 links the CPLANE complex to transition zone architecture.
Vazquez N, Lee C, Valenzuela I, Phan TP, Derderian C, Chávez M, Mooney NA, Demeter J, Aziz-Zanjani MO, Cusco I, Codina M, Martínez-Gil N, Valverde D, Solarat C, Buel AL, Thauvin-Robinet C, Steichen E, Filges I, Joset P, De Geyter J, Vaidyanathan K, Gardner T, Toriyama M, Marcotte EM, Roberson EC, Jackson PK, Reiter JF, Tizzano EF, Wallingford JB. Vazquez N, et al. Among authors: wallingford jb. bioRxiv [Preprint]. 2024 Sep 26:2024.09.25.614984. doi: 10.1101/2024.09.25.614984. bioRxiv. 2024. Update in: Nat Commun. 2025 Jul 1;16(1):5701. doi: 10.1038/s41467-025-61005-8. PMID: 39386566 Free PMC article. Updated. Preprint.
Cilia are essential organelles and variants in genes governing ciliary function result in ciliopathic diseases. ...
Cilia are essential organelles and variants in genes governing ciliary function result in ciliopathic diseases. ...
56 results