journal article
Apr 15, 2017
IFT56 regulates vertebrate developmental patterning by maintaining IFTB complex integrity and ciliary microtubule architecture
Abstract
Cilia are key regulators of animal development and depend on intraflagellar transport (IFT) proteins for their formation and function, yet the roles of individual IFT proteins remain unclear. We examined the Ift56hop mouse mutant and reveal novel insight into the function of IFT56, a poorly understood IFTB protein. Ift56hop mice have normal cilia distribution but display defective cilia structure, including abnormal positioning and number of ciliary microtubule doublets. We show that Ift56hop cilia are unable to accumulate Gli proteins efficiently, resulting in developmental patterning defects in Shh signaling-dependent tissues such as the limb and neural tube. Strikingly, core IFTB proteins are unable to accumulate normally within Ift56hop cilia, including IFT88, IFT81 and IFT27, which are crucial for key processes such as tubulin transport and Shh signaling. IFT56 is required specifically for the IFTB complex, as IFTA components and proteins that rely on IFTA function are unaffected in Ift56hop cilia. These studies define a distinct and novel role for IFT56 in IFTB complex integrity that is crucial for cilia structure and function and, ultimately, animal development.
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Cited By
36
Biochemically validated structural model of the 15‐subunit intraflagellar transport complex IFT‐B
The EMBO Journal
Narcis A Petriman, Marta Loureiro‐López · 2022
Metrics
36
Citations
73
References
Details
- Published
- Apr 15, 2017
- Vol/Issue
- 144(8)
- Pages
- 1544-1553
- License
- View
Authors
Funding
National Institute of General Medical Sciences
Award: T32GM007499
National Science Foundation
Award: DGE-0644492
National Institute of Diabetes and Digestive and Kidney Diseases
Award: P30DK090744
National Institute of Child Health and Human Development
Award: T32HD007149
National Institute of Arthritis and Musculoskeletal and Skin Diseases
Award: R01AR059687
Cite This Article
Daisy Xin, Kasey J. Christopher, Lewie Zeng, et al. (2017). IFT56 regulates vertebrate developmental patterning by maintaining IFTB complex integrity and ciliary microtubule architecture. Development, 144(8), 1544-1553. https://doi.org/10.1242/dev.143255
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