Role of intraflagellar transport protein IFT140 in the formation and function of motile cilia in mammals DOI Creative Commons
Yi Tian Yap, Jiehong Pan, Jian Xu

и другие.

Cellular and Molecular Life Sciences, Год журнала: 2025, Номер 82(1)

Опубликована: Май 10, 2025

Abstract Cilia are microtubular structures extending from the surface of most mammalian cells. They can be categorized as motile cilia and primary sensory cilia. Both types possess intraflagellar transport (IFT) machinery, composed unique protein complexes that travel along microtubules to deliver proteins for ciliary flagellar assembly, disassembly, homeostasis. Although role IFT in formation has been well studied, little is known about its assembly. We generated conditional knockout mice by breeding floxed Ift140 with FOXJ1- Cre transgenic mouse line specifically delete cells assemble Mice deficiency did not have laterality defects or gross; however died prior sexual maturity. Those mutants survived adulthood were completely infertile. Males demonstrated abnormal spermatogenesis associated reduced sperm count motility, together short length flagella, morphology. was diminished epithelial efferent ductules airways. cultured tracheal also had beat frequency (CBF). Ultrastructural studies revealed presence inner outer dynein arms, but an central apparatus, accumulation particles within Overall, localization resulted inadequate function despite proper motor complexes. propose a key assembly certain tissues suggest genetic alterations IFT140 could ciliopathies.

Язык: Английский

Role of intraflagellar transport protein IFT140 in the formation and function of motile cilia in mammals DOI Creative Commons
Yi Tian Yap, Jiehong Pan, Jian Xu

и другие.

Cellular and Molecular Life Sciences, Год журнала: 2025, Номер 82(1)

Опубликована: Май 10, 2025

Abstract Cilia are microtubular structures extending from the surface of most mammalian cells. They can be categorized as motile cilia and primary sensory cilia. Both types possess intraflagellar transport (IFT) machinery, composed unique protein complexes that travel along microtubules to deliver proteins for ciliary flagellar assembly, disassembly, homeostasis. Although role IFT in formation has been well studied, little is known about its assembly. We generated conditional knockout mice by breeding floxed Ift140 with FOXJ1- Cre transgenic mouse line specifically delete cells assemble Mice deficiency did not have laterality defects or gross; however died prior sexual maturity. Those mutants survived adulthood were completely infertile. Males demonstrated abnormal spermatogenesis associated reduced sperm count motility, together short length flagella, morphology. was diminished epithelial efferent ductules airways. cultured tracheal also had beat frequency (CBF). Ultrastructural studies revealed presence inner outer dynein arms, but an central apparatus, accumulation particles within Overall, localization resulted inadequate function despite proper motor complexes. propose a key assembly certain tissues suggest genetic alterations IFT140 could ciliopathies.

Язык: Английский

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