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Related Experiment Video

Updated: Apr 16, 2026

Application of High-speed Super-resolution SPEED Microscopy in Live Primary Cilium

Application of High-speed Super-resolution SPEED Microscopy in Live Primary Cilium

Published on: January 16, 2018

IFT46 plays an essential role in cilia development.

Mi-Sun Lee¹, Kyu-Seok Hwang¹, Hyun-Woo Oh²

¹Department of Biology, Chungnam National University, Daejeon 305-764, Republic of Korea.

Developmental Biology

|February 28, 2015

Summary

Intraflagellar transport 46 (IFT46) is crucial for cilia development in vertebrates. Loss of IFT46 function causes ciliary defects and developmental abnormalities, highlighting its essential role in ciliogenesis.

Keywords:

Cilia Ciliopathy IFT IFT46 Intraflagellar transport KO mouse L/R defect Zebrafish

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Area of Science:

Cell Biology
Developmental Biology
Genetics

Background:

Cilia are vital microtubule-based organelles implicated in numerous human diseases.
Genetic and cellular mechanisms governing ciliogenesis are not fully understood.
Intraflagellar transport (IFT) proteins, including IFT46, are essential for ciliary function.

Purpose of the Study:

To investigate the role of IFT46 in vertebrate ciliary development.
To characterize the function of IFT46 in zebrafish and mouse models.

Main Methods:

Zebrafish ift46 knockdown using morpholino.
Generation and analysis of Ift46 knock-out mice.
Phenotypic analysis of ciliary structure and function, and developmental abnormalities.

Main Results:

ift46 knockdown in zebrafish caused shortened and abnormal cilia in various tissues, leading to ciliopathy-like phenotypes.
Ift46 knockout mice exhibited developmental defects in the brain, neural tube, and heart.
Ift46 mutants showed randomized left-right axis patterning, a critical developmental process.

Conclusions:

IFT46 is essential for proper ciliary development and function in vertebrates.
IFT46 plays a critical role in embryonic development, including organogenesis and left-right axis patterning.
Defects in IFT46 are linked to various human ciliopathies.