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Multidisciplinary Sciences
Girish R. Mali, Ferdos Abid Ali, Clinton K. Lau, Farida Begum, Jerome Boulanger, Jonathan D. Howe, Zhuo A. Chen, Juri Rappsilber, Mark Skehel, Andrew P. Carter
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Limei Wang, Xuecheng Li, Guang Liu, Junmin Pan
Summary: FBB18 plays a crucial role in the assembly of dynein arms by facilitating the folding/stabilization of almost all axonemal dyneins. Mutations in FBB18 result in short or absent cilia with partial loss of dynein arms. FBB18 interacts with multiple DNAAFs simultaneously, indicating the importance of dynein folding/stability in axonemal dynein preassembly.
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Multidisciplinary Sciences
Travis Walton, Hao Wu, Alan Brown
Summary: Axonemal dyneins are tethered to doublet microtubules inside cilia to drive ciliary beating, but the mechanisms regulating their localization and function are poorly understood. Authors report a cryo-EM reconstruction of a three-headed axonemal dynein natively bound to doublet microtubules isolated from cilia, providing a framework to understand the roles of individual subunits.
NATURE COMMUNICATIONS
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Genetics & Heredity
Isabella Aprea, Johanna Raidt, Inga Marlena Hoeben, Niki Tomas Loges, Tabea Noethe-Menchen, Petra Pennekamp, Heike Olbrich, Thomas Kaiser, Luisa Biebach, Frank Tuettelmann, Judit Horvath, Maria Schubert, Claudia Krallmann, Sabine Kliesch, Heymut Omran
Summary: Male fertility issues worldwide may be caused by defects in the preassembly of outer and inner dynein arms, leading to abnormal sperm flagella, impaired sperm motility, and infertility. Studying sperm cells of individuals with mutations in genes related to this process helps understand the clinical relevance of preassembly of dynein arms on male fertility.
Review
Cell Biology
Bryony Braschi, Heymut Omran, George B. Witman, Gregory J. Pazour, K. Kevin Pfister, Elspeth A. Bruford, Stephen M. King
Summary: This review provides an updated consensus nomenclature for components of dynein motors and their assembly factors. Standardizing gene nomenclature facilitates cross-species discussion and genetic comparison. The review also details additional nomenclature updates for vertebrate-specific genes.
JOURNAL OF CELL BIOLOGY
(2022)
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Cell Biology
Stephen M. King
Summary: Axonemal dyneins play a crucial role in powering the beating of motile cilia and flagella. Assembly of these motor complexes requires various cytoplasmic factors, and defects can lead to primary ciliary dyskinesia in mammals. Recent studies suggest that the formation of axonemal dyneins in multiciliated cells may involve liquid-like condensates, but distinguishing this process from other mechanisms can be complex.
JOURNAL OF CELL SCIENCE
(2021)
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Biochemistry & Molecular Biology
Xin Zhu, Jieling Wang, Shufen Li, Karl Lechtreck, Junmin Pan
Summary: This study revealed that the IFT-B protein IFT54 interacts with both kinesin-II and IFT dynein, regulating anterograde transport. Deletions of specific residues in IFT54 resulted in disrupted anterograde trafficking of IFT, with accumulations of IFT motors and complexes in different regions of cilia. This suggests a central role for IFT54 in binding IFT motors during anterograde transport.
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Cell Biology
Amanda J. Smith, Ximena M. Bustamante-Marin, Weining Yin, Patrick R. Sears, Laura E. Herring, Nedyalka N. Dicheva, Francesc Lopez-Giraldez, Shrikant Mane, Robert Tarran, Margaret W. Leigh, Michael R. Knowles, Maimoona A. Zariwala, Lawrence E. Ostrowski
Summary: Mutations in SPAG1, a dynein axonemal assembly factor, cause primary ciliary dyskinesia. This study found that SPAG1 interacts with multiple proteins and plays a role in assembly of dynein arms. A previously uncharacterized SPAG1 isoform was also identified.
JOURNAL OF CELL SCIENCE
(2022)
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Multidisciplinary Sciences
Chiara Scaramuzzino, Emeline C. Cuoc, Patrick Pla, Sandrine Humbert, Frederic Saudou
Summary: In this study, researchers discovered that when a neurotrophin binds to its receptor in neurons, the released calcium ion is sensed by calcineurin, which triggers the retrograde movement of signaling endosomes. This finding suggests that signaling endosomes carry not only their own fuel, but also their own navigational system.
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Cell Biology
Shunya Hiyamizu, Hantian Qiu, Laura Vuolo, Nicola L. Stevenson, Caroline Shak, Kate J. Heesom, Yuki Hamada, Yuta Tsurumi, Shuhei Chiba, Yohei Katoh, David J. Stephens, Kazuhisa Nakayama
Summary: The dynein-2 complex is transported anterogradely within cilia to drive retrograde trafficking of the intraflagellar transport (IFT) machinery containing IFT-A and IFT-B complexes. There are multiple interactions between the dynein-2 and IFT-B subunits, including WDR60 and the DYNC2H1-DYNC2LI1 dimer from dynein-2, and IFT54 and IFT57 from IFT-B. These interactions play a crucial role in the connection between dynein-2 and IFT-B.
JOURNAL OF CELL SCIENCE
(2023)
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Cell Biology
Jin Dai, Gui Zhang, Rama A. Alkhofash, Betlehem Mekonnen, Sahana Saravanan, Bin Xue, Zhen-Chuan Fan, Ewelina Betleja, Douglas G. Cole, Peiwei Liu, Karl Lechtreck
Summary: The loss of Chlamydomonas ARL13 impedes BBSome-dependent protein transport, resulting in overlapping biochemical defects in arl13 and bbs mutant cilia.
JOURNAL OF CELL BIOLOGY
(2022)
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Cell Biology
Ana R. G. De-Castro, Diogo R. M. Rodrigues, Maria J. G. De-Castro, Neide Vieira, Carmen Vieira, Ana X. Carvalho, Reto Gassmann, Carla M. C. Abreu, Tiago J. Dantas
Summary: WDR-60 plays a crucial role in the assembly and functions of cilia by regulating dynein-2 motor complex. Loss of WDR-60 impairs retrograde intraflagellar transport and disrupting the NPHP module partially restores ciliary exit for underpowered retrograde trains in wdr-60 mutants.
JOURNAL OF CELL BIOLOGY
(2021)
Review
Cell Biology
Stephen M. King
Summary: This article discusses the complexity of dynein-driven cilia and the importance of cytosolic factors in the assembly process. It also explores the structural characteristics and potential functions of these factors.
Review
Cell Biology
Dinu Antony, Han G. Brunner, Miriam Schmidts
Summary: The function of cilia is crucial for vertebrate development and health, and dysfunction can lead to various ciliopathies. Major human ciliopathy cases are caused by malfunction of ciliary dynein motor activity, with different types of dynein dysfunction resulting in different disease phenotypes.
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Neurosciences
Yashoda Sharma, Julie S. Jacobs, Elena Sivan-Loukianova, Eugene Lee, Maurice J. Kernan, Daniel F. Eberl
Summary: In this study, the authors characterized the btv gene and found that it encodes the Dync2h1 protein, which is involved in IFT. They also observed morphological and electrophysiological abnormalities in the Johnston's organ and macrochaete bristles of btv mutant flies. Furthermore, they discovered that the NompB protein failed to be cleared from the cilia in the btv mutants. This study highlights the importance of the btv-encoded Dync2h1 in the assembly and function of cilia in different sensory organs.
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