Guardado en:
| Autores principales: | , , , , , , , , , , , , , , , |
|---|---|
| Formato: | Artículo científico |
| Lenguaje: | en |
| Publicado: |
Nature communications
2025
|
| Materias: | |
| Acceso en línea: | https://pubmed.ncbi.nlm.nih.gov/41423487/ |
| Etiquetas: |
Agregar Etiqueta
Sin Etiquetas, Sea el primero en etiquetar este registro!
|
| _version_ | 1868266108338831360 |
|---|---|
| author | Hong, Juyeon Lee, Chanjae Madhu, Gopika Papoulas, Ophelia Atayeter, Ece Hoogerbrugge, Gabriel Pan, Jiehong Takagishi, Maki Manzi, Nadia I Dickinson, Daniel J Horani, Amjad Brody, Steven L Marcotte, Edward M Prakash, Vivek N Park, Tae Joo Wallingford, John B |
| author_facet | Hong, Juyeon Lee, Chanjae Madhu, Gopika Papoulas, Ophelia Atayeter, Ece Hoogerbrugge, Gabriel Pan, Jiehong Takagishi, Maki Manzi, Nadia I Dickinson, Daniel J Horani, Amjad Brody, Steven L Marcotte, Edward M Prakash, Vivek N Park, Tae Joo Wallingford, John B Hong, Juyeon Lee, Chanjae Madhu, Gopika Papoulas, Ophelia Atayeter, Ece Hoogerbrugge, Gabriel Pan, Jiehong Takagishi, Maki Manzi, Nadia I Dickinson, Daniel J Horani, Amjad Brody, Steven L Marcotte, Edward M Prakash, Vivek N Park, Tae Joo Wallingford, John B |
| collection | PubMed - marine biology |
| contents | A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function. Hong, Juyeon Lee, Chanjae Madhu, Gopika Papoulas, Ophelia Atayeter, Ece Hoogerbrugge, Gabriel Pan, Jiehong Takagishi, Maki Manzi, Nadia I Dickinson, Daniel J Horani, Amjad Brody, Steven L Marcotte, Edward M Prakash, Vivek N Park, Tae Joo Wallingford, John B Cilia Animals Mice Humans Microtubules Xenopus laevis Multiprotein Complexes The beating of cilia on multi-ciliated cells (MCCs) is essential for normal development and homeostasis in animals. But while the structure and function of basal bodies and axonemes have received significant attention recently, the distal tips of MCC cilia remain relatively poorly defined. Here, we characterize the molecular organization of the distal tip of vertebrate MCC cilia, characterizing two distinct domains occupied by distinct protein constituents. Using frog, mouse, and human MCCs, we find that two largely uncharacterized proteins, Ccdc78 and Ccdc33, occupy a previously undefined region at the extreme distal tip, and these are required for the normal organization of all other known tip proteins. Ccdc78 and Ccdc33 each display robust microtubule-bundling activity both in vivo and in vitro, yet each is independently required for normal length regulation of MCC cilia. Moreover, loss of each protein elicits a distinct pattern of defective cilia beating and resultant fluid flow. Thus, two previously undefined proteins form a key module essential for organizing and stabilizing the distal tip of motile cilia in vertebrate MCCs. We propose that these ill-defined proteins represent potential disease loci for motile ciliopathies. |
| format | Artículo científico |
| id | pubmed_41423487 |
| institution | PubMed |
| language | en |
| publishDate | 2025 |
| publisher | Nature communications |
| record_format | pubmed |
| spellingShingle | A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function. Hong, Juyeon Lee, Chanjae Madhu, Gopika Papoulas, Ophelia Atayeter, Ece Hoogerbrugge, Gabriel Pan, Jiehong Takagishi, Maki Manzi, Nadia I Dickinson, Daniel J Horani, Amjad Brody, Steven L Marcotte, Edward M Prakash, Vivek N Park, Tae Joo Wallingford, John B Cilia Animals Mice Humans Microtubules Xenopus laevis Multiprotein Complexes A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function. Hong, Juyeon Lee, Chanjae Madhu, Gopika Papoulas, Ophelia Atayeter, Ece Hoogerbrugge, Gabriel Pan, Jiehong Takagishi, Maki Manzi, Nadia I Dickinson, Daniel J Horani, Amjad Brody, Steven L Marcotte, Edward M Prakash, Vivek N Park, Tae Joo Wallingford, John B Cilia Animals Mice Humans Microtubules Xenopus laevis Multiprotein Complexes The beating of cilia on multi-ciliated cells (MCCs) is essential for normal development and homeostasis in animals. But while the structure and function of basal bodies and axonemes have received significant attention recently, the distal tips of MCC cilia remain relatively poorly defined. Here, we characterize the molecular organization of the distal tip of vertebrate MCC cilia, characterizing two distinct domains occupied by distinct protein constituents. Using frog, mouse, and human MCCs, we find that two largely uncharacterized proteins, Ccdc78 and Ccdc33, occupy a previously undefined region at the extreme distal tip, and these are required for the normal organization of all other known tip proteins. Ccdc78 and Ccdc33 each display robust microtubule-bundling activity both in vivo and in vitro, yet each is independently required for normal length regulation of MCC cilia. Moreover, loss of each protein elicits a distinct pattern of defective cilia beating and resultant fluid flow. Thus, two previously undefined proteins form a key module essential for organizing and stabilizing the distal tip of motile cilia in vertebrate MCCs. We propose that these ill-defined proteins represent potential disease loci for motile ciliopathies. |
| title | A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function. |
| topic | Cilia Animals Mice Humans Microtubules Xenopus laevis Multiprotein Complexes |
| url | https://pubmed.ncbi.nlm.nih.gov/41423487/ |