Cilia are key regulators of animal development and depend on intraflagellar transport (IFT) proteins to form and function, yet the roles of individual IFTs are still 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. Furthermore, 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, we further demonstrate that core IFTB proteins are unable to accumulate normally within Ift56hop cilia, including IFT88, IFT81, and IFT27, which are critical 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.
IFT56 regulates vertebrate developmental patterning by maintaining IFTB integrity and ciliary microtubule architecture
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