IFT46 plays crucial roles in craniofacial and cilia development

Park, Inji; Lee, Hyun-Kyung; Kim, Chowon; Ismail, Tayaba; Kim, Yoo-Kyung; Park, Jeen‐Woo; Kwon, Oh Nam; Kang, Beom Sik; Lee, Dong‐Seok; Park, Tae-Joo; Park, Mae-Ja; Choi, Sun‐Cheol; Lee, Hyun‐Shik

doi:10.1016/j.bbrc.2016.06.083

Cited by 11 publications

(10 citation statements)

References 34 publications

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“…It binds IFT52 and IFT88 to form a stable trimetric complex 18 . Research on Chlamydomonas reinhardtii , Caenorhabditis elegans and mammals showed that IFT46 is essential for cilia assembly and vertebrate development 19 , 20 . In addition, a study on C .…”

Section: Introductionmentioning

confidence: 99%

Intraflagellar transport 46 (IFT46) is essential for trafficking IFT proteins between cilia and cytoplasm in Paramecium

Shi

Shen

2018

Sci Rep

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Intraflagellar transport (IFT) is a bi-directional process by which particles are carried within the cilia or flagella. This process is essential for ciliary growth and functional maintenance. The IFT complex B (IFTB) is linked to a kinesin motor for anterograde transport towards the ciliary tip. The IFT complex A (IFTA) is connected to a dynein motor for retrograde transport towards the ciliary basis. This study focuses on IFT46, an IFTB member that participates in this process. In Paramecium, a GFP-labelled IFT46 protein was found in basal bodies and in some cilia, mostly those undergoing biogenesis. RNA interference against IFT46 in Paramecium triggered severe defects in ciliary growth and architecture, including a decreased cilia number and shortened cilia length. This result differed from that obtained from the cells that were depleted of IFT80, another IFTB protein. Moreover, IFT57-GFP fusion protein abnormally accumulated in the cortex and cytoplasm in IFT46-depleted cells compared with the control. Furthermore, transcriptomic analysis showed that IFT46 depletion induced the abnormal expression of several genes that encodeding kinesin and dynein chains. These findings together indicate that IFT46 plays important roles in trafficking IFT proteins between the cytoplasm and cilia of Paramecium.

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Section: Introductionmentioning

confidence: 99%

Intraflagellar transport 46 (IFT46) is essential for trafficking IFT proteins between cilia and cytoplasm in Paramecium

Shi

Shen

2018

Sci Rep

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“…IFT46 is another component of the IFT-B complex. Park et al (2016) reported that knockdown of IFT46 using morpholino microinjections results in a loss of primary cilia and defective craniofacial development. Their findings further suggest that IFT46 regulates ciliogenesis, neurogenesis, and craniofacial development by modulating Wnt/PCP and Shh signaling pathways (Park et al 2016).…”

Section: Primary Cilia Regulate Cartilage Formationmentioning

confidence: 99%

“…Park et al (2016) reported that knockdown of IFT46 using morpholino microinjections results in a loss of primary cilia and defective craniofacial development. Their findings further suggest that IFT46 regulates ciliogenesis, neurogenesis, and craniofacial development by modulating Wnt/PCP and Shh signaling pathways (Park et al 2016). In addition to IFT complex B components, IFT motor kinesin family proteins such as KIF3A and KIF7 have been reported to play an important role in chondrocyte proliferation and differentiation and cartilage development (reviewed by .…”

Section: Primary Cilia Regulate Cartilage Formationmentioning

confidence: 99%

Role of Primary Cilia in Bone and Cartilage

et al. 2021

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The primary cilium is a nonmotile microtubule-based organelle in most vertebrate cell types. The primary cilium plays a critical role in tissue development and homeostasis by sensing and transducing various signaling pathways. Ciliary proteins such as intraflagellar transport (IFT) proteins as well as ciliary motor proteins, kinesin and dynein, comprise a bidirectional intraflagellar transport system needed for cilia formation and function. Mutations in ciliary proteins that lead to loss or dysfunction of primary cilia cause ciliopathies such as Jeune syndrome and Ellis–van Creveld syndrome and cause abnormalities in tooth development. These diseases exhibit severe skeletal and craniofacial dysplasia, highlighting the significance of primary cilia in skeletal development. Cilia are necessary for the propagation of hedgehog, transforming growth factor β, platelet-derived growth factor, and fibroblast growth factor signaling during osteogenesis and chondrogenesis. Ablation of ciliary proteins such as IFT80 or IFT20 blocks cilia formation, which inhibits osteoblast differentiation, osteoblast polarity, and alignment and reduces bone formation. Similarly, cilia facilitate chondrocyte differentiation and production of a cartilage matrix. Cilia also play a key role in mechanosensing and are needed for increased bone formation in response to mechanical forces.

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“…The intraflagella transport (IFT) IFT46 gene is thought to be associated with regulation and maintenance of ciliary length during ciliogenesis [ 39 ]. Lara Millán presented a study measuring ciliary length in 27 PCD patients’ samples in relation to high-speed video microscopy (HSVM) and TEM data, and IFT46 , FOXJ1 and DNAI2 gene expression.…”

Section: Oral Presentationsmentioning

confidence: 99%

Proceedings of the 2nd BEAT-PCD conference and 3rd PCD training school: part 1

et al. 2018

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Primary ciliary dyskinesia (PCD) is a rare heterogenous condition that causes progressive suppurative lung disease, chronic rhinosinusitis, chronic otitis media, infertility and abnormal situs. ‘Better Experimental Approaches to Treat Primary Ciliary Dyskinesia’ (BEAT-PCD) is a network of scientists and clinicians coordinating research from basic science through to clinical care with the intention of developing treatments and diagnostics that lead to improved long-term outcomes for patients. BEAT-PCD activities are supported by EU funded COST Action (BM1407). The second BEAT-PCD conference, and third PCD training school were held jointly in April 2017 in Valencia, Spain. Presentations and workshops focussed on advancing the knowledge and skills relating to PCD in: basic science, epidemiology, diagnostic testing, clinical management and clinical trials. The multidisciplinary conference provided an interactive platform for exchanging ideas through a program of lectures, poster presentations, breakout sessions and workshops. Three working groups met to plan consensus statements. Progress with BEAT-PCD projects was shared and new collaborations were fostered. In this report, we summarize the meeting, highlighting developments made during the meeting.Electronic supplementary materialThe online version of this article (10.1186/s12919-018-0098-9) contains supplementary material, which is available to authorized users.

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IFT46 plays crucial roles in craniofacial and cilia development

Cited by 11 publications

References 34 publications

Intraflagellar transport 46 (IFT46) is essential for trafficking IFT proteins between cilia and cytoplasm in Paramecium

Intraflagellar transport 46 (IFT46) is essential for trafficking IFT proteins between cilia and cytoplasm in Paramecium

Role of Primary Cilia in Bone and Cartilage

Proceedings of the 2nd BEAT-PCD conference and 3rd PCD training school: part 1

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