IFT46 plays crucial roles in craniofacial and cilia development.
The intraflagellar transport (IFT) system is essential for bidirectional movement of ciliary components from the basal body to the tip beneath the ciliary sheath and is conserved for cilia and flagella formation in most vertebrates. IFT complex A is involved in anterograde trafficking, whereas complex B is involved in retrograde trafficking. IFT46 is well known as a crucial component of IFT complex B, however, its developmental functions are poorly understood. In this study, we investigated the novel functions of IFT46 during vertebrate development, especially, ciliogenesis and neurogenesis, because IFT46 is strongly expressed in both multiciliated cells of epithelial and neural tissues. Knockdown of IFT46 using morpholino microinjections caused shortening of the body axis as well as the formation of fewer and shorter cilia. Furthermore, loss of IFT46 down-regulated the expression of the neural plate and neural tube markers, thus may influence Wnt/planar cell polarity and the sonic hedgehog signaling pathway during neurogenesis. In addition, loss of IFT46 caused craniofacial defects by interfering with cartilage formation. In conclusion, our results depict that IFT46 plays important roles in cilia as well as in neural and craniofacial development.
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Inji
I
Park
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Hyun-Kyung
HK
Lee
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Chowon
C
Kim
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Tayaba
T
Ismail
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Yoo-Kyung
YK
Kim
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Jeen-Woo
JW
Park
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Oh-Shin
OS
Kwon
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Beom Sik
BS
Kang
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea.
Dong-Seok
DS
Lee
School of Life Sciences, Ulsan National Institute of Science and Technology (UNIST), Ulsan, 44919, South Korea.
Tae-Joo
TJ
Park
Department of Anatomy, College of Medicine, Kyungpook National University, Daegu, 41944, South Korea.
Mae-Ja
MJ
Park
Department of Biomedical Sciences, University of Ulsan, College of Medicine, Seoul, 05505, South Korea. Electronic address: choisc@amc.seoul.kr.
Sun-Cheol
SC
Choi
ABRC, CMRI, School of Life Sciences, BK21 Plus KNU Creative BioResearch Group, Kyungpook National University, Daegu, 41566, South Korea. Electronic address: leeh@knu.ac.kr.
Hyun-Shik
HS
Lee
D000818:Animals; D002923:Cilia; D005145:Face / Q000196:embryology*; D047908:Intracellular Signaling Peptides and Proteins / Q000502:physiology*; D012886:Skull / Q000196:embryology*; D014981:Xenopus / Q000196:embryology*
D047908:Intracellular Signaling Peptides and Proteins
Ciliogenesis; Craniofacial development; IFT46; Neurogenesis