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Canonical and noncanonical intraflagellar transport regulates craniofacial skeletal development

2016; National Academy of Sciences; Volume: 113; Issue: 19 Linguagem: Inglês

10.1073/pnas.1519458113

1091-6490

Kazuo Noda, Megumi Kitami, Kohei Kitami, Masaru Kaku, Yoshihiro Komatsu,

Renal and related cancers

Significance Intraflagellar transport (IFT) plays a critical role in assembling primary cilia that mediate growth factor signaling. The disruption or dysfunction of IFT components can generate multiple diseases, including skeletal dysplasia. However, the mechanism by which IFT regulates skeletogenesis remains elusive. In the present study, we show that neural crest-specific deletion of the gene that encodes intraflagellar transport 20 (IFT20) in mice compromises ciliogenesis and the intracellular transport of collagen, leading to osteopenia in the face. Our findings highlight a unique function of IFT beyond its role in cilium assembly during craniofacial development, suggesting that IFT20 is indispensable for the regulation of not only ciliogenesis but also the intracellular trafficking of collagen in the unique multipotent stem cell population of cranial neural crests.