The kinesin-4 protein Kif7 regulates mammalian Hedgehog signalling by organizing the cilium tip compartment
Open accessAbstract
Mammalian Hedgehog (Hh) signal transduction requires a primary cilium, a microtubule-based organelle, and the Gli-Sufu complexes that mediate Hh signalling, which are enriched at cilia tips. Kif7, a kinesin-4 family protein, is a conserved regulator of the Hh signalling pathway and a human ciliopathy protein. Here we show that Kif7 localizes to the cilium tip, the site of microtubule plus ends, where it limits cilium length and controls cilium structure. Purified recombinant Kif7 binds the plus ends of growing microtubules in vitro, where it reduces the rate of microtubule growth and increases the frequency of microtubule catastrophe. Kif7 is not required for normal intraflagellar transport or for trafficking of Hh pathway proteins into cilia. Instead, a central function of Kif7 in the mammalian Hh pathway is to control cilium architecture and to create a single cilium tip compartment, where Gli-Sufu activity can be correctly regulated.
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Beta-arrestins are recruited to the proximal C-terminus of GPR161 in a GRK2-dependent manner.
“β-arrestins are adaptor proteins that are recruited to the proximal C-terminus of GPR161 in a manner dependent on G protein-coupled receptor kinase 2 (GRK2)138.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [138]
The Results sections on KIF7 at growing microtubule plus-ends and ciliary tip organization discuss KIF7, microtubules, and Gli2, not GPR161 or β-arrestins (page unavailable). Searches for GPR161, β-arrestin recruitment, and GRK2 found no passage addressing the claimed interaction. (LLM, conf=0.76)
Experimental — accuracy not yet measuredGPR161 is removed from primary cilia in a SMO-dependent and beta-arrestin-dependent manner after Hedgehog pathway activation.
“GPR161 is also removed from primary cilia in a SMO-dependent and β-arrestin-dependent manner following activation of HH signalling 28, 138.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [138]
The Results section on Gli2 and Sufu localization reports that Smoothened moves into cilia after pathway stimulation, but does not address GPR161 leaving cilia. Searches for GPR161, ciliary exit, and β-arrestin found no passage in our copy supporting the claimed dependencies. (LLM, conf=0.76)
Experimental — accuracy not yet measured
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Developmental and regenerative paradigms of cilia regulated hedgehog signaling
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“Multiple ciliary tip-like compartments form in the absence of Kif7 [88], and Kif7 not only recognizes but also stabilizes a GTP-form of tubulin to promote its own microtubule-end localization [90] (Figure 1C).”
BackgroundInfluentialNeurodevelopmental origins of bipolar disorder: iPSC models
O’Shea, K. S., & McInnis, M. G. (2015) · Molecular and Cellular Neuroscience · doi:10.1016/j.mcn.2015.11.006 · Cited by 107 · see citing works
Mixed signals from the cell's antennae: primary cilia in cancer
Eguether, T., & Hahne, M. (2018) · EMBO Reports · doi:10.15252/embr.201846589 · Cited by 100 · see citing works
“The transcription factors of the pathway (Gli1, Gli2 and Gli3) are transported to the tip of the cilium in a complex with SuFu and Kif7 [26,31,32].”
Cellular Mechanisms of Ciliary Length Control
Keeling, J., Tsiokas, L., & Maskey, D. (2016) · Cells · doi:10.3390/cells5010006 · Cited by 97 · see citing works
“Kif7 has recently been reported to localize to the ciliary tip and to modulate hedgehog signaling at least partially by inducing tubulin depolymerization [12].”
BackgroundAssembly, Functions and Evolution of Archaella, Flagella and Cilia
Khan, S., & Scholey, J. M. (2018) · Current Biology · doi:10.1016/j.cub.2018.01.085 · Cited by 96 · see citing works
Zhang, J., Fan, J., Zeng, X. L., Nie, M., Luan, J., Wang, Y., Ju, D., & Yin, K. (2020) · Acta Pharmaceutica Sinica B · doi:10.1016/j.apsb.2020.10.022 · Cited by 92 · see citing works
Mechanism and regulation of kinesin motors.
Yildiz, A. (2025) · Nature reviews. Molecular cell biology · doi:10.1038/s41580-024-00780-6 · Cited by 88 · see citing works
Autophagy and regulation of cilia function and assembly
Orhon, I., Dupont, N., Pampliega, O., Cuervo, A. M., & Codogno, P. (2014) · Cell Death and Differentiation · doi:10.1038/cdd.2014.171 · Cited by 85 · see citing works
“the cilium creates a specialized compartment at the top of the cilia where the activity of the Gli proteins is regulated.(30) The”
BackgroundWeghe, J. C. V. D., Rusterholz, T. D. S., Latour, B. L., Grout, M. E., Aldinger, K. A., Shaheen, R., Dempsey, J. C., Maddirevula, S., Cheng, Y.-H. H., Phelps, I. G., et al. (2017) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2017.05.010 · Cited by 84 · see citing works
Subcutaneous and Visceral Adipose-Derived Mesenchymal Stem Cells: Commonality and Diversity
Ritter, A., Friemel, A., Roth, S., Kreis, N., Hoock, S. C., Safdar, B. K., Fischer, K., Möllmann, C. J., Solbach, C., Louwen, F., et al. (2019) · Cells · doi:10.3390/cells8101288 · Cited by 80 · see citing works
“Fluorescent intensity was measured while using line-scan-based analysis in ImageJ (National Institutes of Health), as described [15,18].”
MethodDalbay, M. T., Thorpe, S. D., Connelly, J. T., Chapple, J. P., & Knight, M. M. (2015) · Stem Cells · doi:10.1002/stem.1975 · Cited by 77 · see citing works
Dysfunction of the ciliary ARMC9/TOGARAM1 protein module causes Joubert syndrome
Latour, B. L., Van De Weghe, J. C., Rusterholz, T. D. S., Letteboer, S. J. F., Gomez, A., Shaheen, R., Gesemann, M., Karamzade, A., Asadollahi, M., Barroso-Gil, M., et al. (2020) · Journal of Clinical Investigation · doi:10.1172/jci131656 · Cited by 76 · see citing works
“This is in line with our findings of decreased axonemal microtubule acetylation and stability with ARMC9 and TOGARAM1 dysfunction, as well as previously published findings with Kif7 and Armc9 dysfunction (33, 68).”
BackgroundResultInfluentialDynarrestin, a Novel Inhibitor of Cytoplasmic Dynein
Höing, S., Yeh, T., Baumann, M., Martinez, N. E., Habenberger, P., Kremer, L., Drexler, H. C. A., Küchler, P., Reinhardt, P., Choidas, A., et al. (2018) · Cell chemical biology · doi:10.1016/j.chembiol.2017.12.014 · Cited by 76 · see citing works
“For live imaging of IFT movement in primary cilia, 107 mouse IMCD3 cells were resuspended in 0.5 ml OPTI-MEM and electroporated with 10 μg plasmid DNAs (IFT88– GFP and ARL13b–mCherry in a pCAG-based expression vector, kindly provided by Kathryn Anderson (He et al., 2014) at 240 V using a BTX electro cell manipulator 6…”
MethodInfluentialPrimary Cilia Are Dysfunctional in Obese Adipose-Derived Mesenchymal Stem Cells
Ritter, A., Friemel, A., Kreis, N., Hoock, S. C., Roth, S., Kielland-Kaisen, U., Brüggmann, D., Solbach, C., Louwen, F., & Yuan, J. (2018) · Stem Cell Reports · doi:10.1016/j.stemcr.2017.12.022 · Cited by 73 · see citing works
“…axonemal MTs, a line-scan analysis of fluorescent acetylated a-tubulinwas performed from the proximal base to the distal tip of cilia in stained ASCs normalized to the cilium length marked by Arl13b staining in visceral (Figure 2A) and subcutaneous ASCs (Figure 2B), as described (He et al., 2014).”
Ghiretti, A. E., Thies, E., Tokito, M., Lin, T., Ostap, E., Kneussel, M., & Holzbaur, E. (2016) · Neuron · doi:10.1016/j.neuron.2016.10.003 · Cited by 62 · see citing works
“Other members of the kinesin-4 family, including KIF4A and KIF21A, have been implicated in the regulation of microtubule dynamics (He et al., 2014; van der Vaart et al., 2013), so we investigated a possible role for KIF21B in the regulation of themicrotubule cytoskeleton in dendrites using end…”
InfluentialRoutes and machinery of primary cilium biogenesis
Bernabé‐Rubio, M., & Alonso, M. A. (2017) · Cellular and Molecular Life Sciences · doi:10.1007/s00018-017-2570-5 · Cited by 72 · see citing works
“Kif7, a kinesin-4 family protein that is a conserved regulator of the Hh signaling pathway and a human ciliopathy protein, binds to the distal end of axonemal microtubules and organizes a specialized compartment where the activity of the Gli family of Hh transcription factors is regulated [30, 74, 75].”
BackgroundInfluentialMicrotubule Motors Drive Hedgehog Signaling in Primary Cilia
He, M., Agbu, S. O., & Anderson, K. V. (2016) · Trends in Cell Biology · doi:10.1016/j.tcb.2016.09.010 · Cited by 69 · see citing works
“The primary cilia of cultured fibroblasts carrying either mouse and human Kif7 mutations are longer and less stable than wild type control, which demonstrates that KIF7 plays a role in regulating cilia structure [99,100].”
BackgroundInfluentialSelf-citationHedgehog Signaling: Implications in Cancers and Viral Infections
Iriana, S., Asha, K., Repak, M., & Sharma-Walia, N. (2021) · International Journal of Molecular Sciences · doi:10.3390/ijms22031042 · Cited by 73 · see citing works
“KIF7 must relay the signals from the membrane protein SMO to the GLI family transcription factors [38].”
BackgroundMeasuring the primary cilium length: improved method for unbiased high-throughput analysis
Dummer, A., Poelma, C., DeRuiter, M. C., Goumans, M.-J. T. H., & Hierck, B. P. (2016) · Cilia · doi:10.1186/s13630-016-0028-2 · Cited by 73 · see citing works
“Along the axoneme, intraflagellar transport (IFT) regulates the availability of proteins for signal transduction, such as members of the Hh signaling cascade [9] and regulates the availability of building blocks for the axoneme to establish the cilium [10].”
BackgroundArl3 and RP2 regulate the trafficking of ciliary tip kinesins
Schwarz, N., Lane, A., Jovanovic, K., Parfitt, D. A., Aguilà, M., Thompson, C. L., da Cruz, L., Coffey, P. J., Chapple, J. P., Hardcastle, A. J., et al. (2017) · Human Molecular Genetics · doi:10.1093/hmg/ddx143 · Cited by 71 · see citing works
“structure and organisation of primary cilia tips (23).”
BackgroundResultInfluentialEngelke, M. F., Waas, B., Kearns, S. E., Suber, A., Boss, A. P., Allen, B. L., & Verhey, K. J. (2019) · Current Biology · doi:10.1016/j.cub.2019.02.043 · Cited by 69 · see citing works
Dynein and intraflagellar transport
Hou, Y., & Witman, G. B. (2015) · Experimental Cell Research · doi:10.1016/j.yexcr.2015.02.017 · Cited by 70 · see citing works
Deciphering the role of hedgehog signaling in pancreatic cancer
Gu, D., Kelly, E., & Xie, J. (2016) · Journal of Biomedical Research · doi:10.7555/jbr.30.20150107 · Cited by 70 · see citing works
“Numerous studies indicate a critical role of primary cilium for Hh signal transduction[4-7].”
BackgroundRamsbottom, S. A., Molinari, E., Srivastava, S., Silberman, F., Henry, C., Alkanderi, S., Devlin, L. A., White, K., STEEL, D. H., Saunier, S., et al. (2018) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1809432115 · Cited by 70 · see citing works
“It is intriguing to note that elongated cilia have also been found in a wide range of other ciliopathy models including: Bbs-4 null mice (18), jckmice (19), Meckel syndrome (20), Kif7 mutated cells (21), and JBTS secondary to KIAA0556 mutations (22), suggesting elongated primary cilia may be a widespread feature of ci…”
BackgroundThe Joubert–Meckel–Nephronophthisis Spectrum of Ciliopathies
Weghe, J. C. V. D., Gómez, A. E., & Doherty, D. (2022) · Annual Review of Genomics and Human Genetics · doi:10.1146/annurev-genom-121321-093528 · Cited by 67 · see citing works
Seriously cilia: A tiny organelle illuminates evolution, disease, and intercellular communication
Derderian, C., Canales, G., & Reiter, J. F. (2023) · Developmental Cell · doi:10.1016/j.devcel.2023.06.013 · Cited by 69 · see citing works
Lithium chloride modulates chondrocyte primary cilia and inhibits Hedgehog signaling
Thompson, C. L., Wiles, A. K., Poole, C. A., & Knight, M. M. (2015) · The FASEB Journal · doi:10.1096/fj.15-274944 · Cited by 68 · see citing works
Srivastava, S., Ramsbottom, S. A., Molinari, E., Alkanderi, S., Filby, A. J., White, K., Henry, C., Saunier, S., Miles, C. G., & Sayer, J. A. (2017) · Human Molecular Genetics · doi:10.1093/hmg/ddx347 · Cited by 69 · see citing works
“Cells depleted of KIF7 (JBTSF12) and KIAA0556 (JBTS26) also have extended axonemes (34,35).”
BackgroundProteins that control the geometry of microtubules at the ends of cilia
Louka, P., Vasudevan, K. K., Guha, M., Joachimiak, E., Włoga, D., Tomasi, R. F.-X., Baroud, C. N., Dupuis‐Williams, P., Galati, D. F., Pearson, C. G., et al. (2018) · The Journal of Cell Biology · doi:10.1083/jcb.201804141 · Cited by 68 · see citing works
“KIF7 may contribute to the distal segment organization by shortening the ends of axonemal microtubules (He et al., 2014).”
BackgroundInfluentialCilia and polycystic kidney disease
Ma, M. (2020) · Seminars in Cell and Developmental Biology · doi:10.1016/j.semcdb.2020.05.003 · Cited by 70 · see citing works
Regulation of Hedgehog Signalling Inside and Outside the Cell
Ramsbottom, S. A., & Pownall, M. E. (2016) · Journal of Developmental Biology · doi:10.3390/jdb4030023 · Cited by 66 · see citing works
“This microtubule regulation generates the compartment at the ciliary tip, allowing components of the hedgehog pathway to be appropriately localised and processed [179].”
BackgroundKIAA0556 is a novel ciliary basal body component mutated in Joubert syndrome
Sanders, A. A. W. M., de Vrieze, E., Alazami, A. M., Alzahrani, F., Malarkey, E. B., Sorusch, N., Tebbe, L., Kuhns, S., van Dam, T. J. P., Alhashem, A. M., et al. (2015) · Genome biology · doi:10.1186/s13059-015-0858-z · Cited by 65 · see citing works
“and associated with hydrolethalus syndrome, binds to MT plus ends and regulates MT catastrophe and B-tubule integrity at the ciliary tip [20].”
BackgroundArveseth, C. D., Happ, J. T., Hedeen, D. S., Zhu, J., Capener, J. L., Shaw, D. K., Deshpande, I., Liang, J., Xu, J., Stubben, S. L., et al. (2021) · PLoS Biology · doi:10.1371/journal.pbio.3001191 · Cited by 60 · see citing works
The Hedgehog Signaling Pathway in Idiopathic Pulmonary Fibrosis: Resurrection Time
Effendi, W. I., & Nagano, T. (2021) · International Journal of Molecular Sciences · doi:10.3390/ijms23010171 · Cited by 59 · see citing works
“The core components that mediate the Hh signal response in vertebrae are two patched receptors (Ptch1, Ptch2), a key signal transducer smoothened receptor (Smo), three glioma-associated oncogene (Gli) transcription factors (Gli1, Gli2, Gli3), the suppressed fusion homolog (Sufu), and kinesine protein 7 (Kif7) [14].”
BackgroundInfluentialThe multifaceted roles of primary cilia in the development of the cerebral cortex
Hasenpusch-Theil, K., & Theil, T. (2021) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2021.630161 · Cited by 66 · see citing works
“Primary cilia are pivotal to interneuron migration since disrupting ciliary functions in cortical interneurons interfered with the migratory process and led to abnormal positioning of interneurons in the adult cortex (Baudoin et al., 2012; Higginbotham et al., 2012).”
BackgroundKinesin-4 KIF21B is a potent microtubule pausing factor
van Riel, W. E., Rai, A., Bianchi, S., Katrukha, E. A., Liu, Q., Heck, A. J. R., Hoogenraad, C. C., Steinmetz, M. O., Kapitein, L. C., & Akhmanova, A. S. (2017) · eLife · doi:10.7554/elife.24746 · Cited by 64 · see citing works
“Another kinesin-4 family member, KIF7, is immotile; it participates in organizing the tips of ciliary MTs by reducing the MT growth rate and promoting catastrophes (He et al., 2014).”
The hedgehog pathway and ocular developmental anomalies
Cavodeassi, F., Creuzet, S., & Etchevers, H. (2018) · Human Genetics · doi:10.1007/s00439-018-1918-8 · Cited by 64 · see citing works
Shortened primary cilium length and dysregulated Sonic hedgehog signaling in Niemann-Pick C1 disease
Canterini, S., Dragotto, J., Dardis, A., Zampieri, S., Stefano, M. E. D., Mangia, F., Erickson, R. P., & Fiorenza, M. T. (2017) · Human Molecular Genetics · doi:10.1093/hmg/ddx118 · Cited by 63 · see citing works
Mori, R. D., Romani, M., D’Arrigo, S., Zaki, M. S., Lorefice, E., Tardivo, S., Biagini, T., Stanley, V., Musaev, D., Fluss, J., et al. (2017) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2017.08.017 · Cited by 62 · see citing works
“In the absence of functional KIF7, the ciliary tip compartment becomes disorganized, leading to the formation of ectopic tip-like compartments where GLI-SUFU complexes localize and are inappropriately activated even in the absence of SHH ligand.(52) The same constellation of cranio-facial abnormalities along with deve…”
BackgroundHarris, J. A., Liu, Y., Yang, P., Kner, P., & Lechtreck, K. F. (2015) · Molecular Biology of the Cell · doi:10.1091/mbc.e15-08-0608 · Cited by 60 · see citing works
Control of meiotic chromosomal bouquet and germ cell morphogenesis by the zygotene cilium
Mytlis, A., Kumar, V., Qiu, T., Deis, R., Hart, N., Levy, K., Masek, M., Shawahny, A., Ahmad, A., Eitan, H., et al. (2022) · Science · doi:10.1126/science.abh3104 · Cited by 60 · see citing works
The intraflagellar transport cycle
Lacey, S. E., & Pigino, G. (2024) · Nature Reviews Molecular Cell Biology · doi:10.1038/s41580-024-00797-x · Cited by 64 · see citing works
“Once at the tip, KIF7 establishes a specialized signalling compartment 26,156,158 that results in activation of GLI2, which can elicit a transcriptional response back in the cell body 12 .”
Liu, Y. C., Couzens, A. L., Deshwar, A. R., McBroom, L. D. B., Zhang, X., Puviindran, V., Scott, I. C., Gingras, A., Hui, C., & Angers, S. (2014) · Science Signaling · doi:10.1126/scisignal.2005608 · Cited by 59 · see citing works
Cilia Distal Domain: Diversity in Evolutionarily Conserved Structures
Soares, H., Carmona, B., Nolasco, S., Melo, L. V., & Gonçalves, J. (2019) · Cells · doi:10.3390/cells8020160 · Cited by 59 · see citing works
“KIF7 displays no motility, is unable to diffuse along microtubules [199], localizes to the base of primary cilium in the absence of Hh signaling, and shows an Hh depending-accumulation at the ciliary tip [198].”
InfluentialThe developmental biology of kinesins
Konjikusic, M. J., Gray, R. S., & Wallingford, J. B. (2020) · Developmental Biology · doi:10.1016/j.ydbio.2020.09.009 · Cited by 57 · see citing works
The role of Hedgehog and Notch signaling pathway in cancer
Xia, R., Xu, M., Yang, J., & Ma, X. (2022) · Molecular Biomedicine · doi:10.1186/s43556-022-00099-8 · Cited by 57 · see citing works
Kinesin superfamily proteins and the regulation of microtubule dynamics in morphogenesis
Niwa, S. (2014) · Anatomical Science International · doi:10.1007/s12565-014-0259-5 · Cited by 56 · see citing works
“Actually, a recent study has shown that KIF7, a member of Kinesin-4, regulates the length of primary cilia (He et al. 2014).”
BackgroundTaylor, S. P., Bosakova, M. K., Vařecha, M., Bálek, L., Bárta, T., Trantı́rek, L., Jelínková, I., Durán, I., Veselá, I., Forlenza, K. N., et al. (2016) · Human Molecular Genetics · doi:10.1093/hmg/ddw240 · Cited by 55 · see citing works
“twisted, similar to the structures of cilia from Kif7 mutants Kif7 and Kif7 (39), suggesting that ICK may be required for ciliary structural integrity.”
BackgroundBowie, E., Norris, R., Anderson, K. V., & Goetz, S. C. (2018) · PLoS Genetics · doi:10.1371/journal.pgen.1007844 · Cited by 55 · see citing works
Self-citationRab34 GTPase mediates ciliary membrane formation in the intracellular ciliogenesis pathway
Ganga, A. K., Kennedy, M. C., Oguchi, M. E., Gray, S. D., Oliver, K. E., Knight, T. A., Cruz, E. M. D. L., Homma, Y., Fukuda, M., & Breslow, D. K. (2021) · Current Biology · doi:10.1016/j.cub.2021.04.075 · Cited by 53 · see citing works
Schwarz, T., Prieler, B. E. M., Schmid, J. A., Grzmil, P., & Neesen, J. (2017) · European Journal of Cell Biology · doi:10.1016/j.ejcb.2017.02.003 · Cited by 53 · see citing works
“For the induction of cilia formation in NIH 3T3 cells confluent cultures were serum starved for 24 h prior further analysis (He et al., 2014).”
MethodKinesin-4 KIF21B limits microtubule growth to allow rapid centrosome polarization in T cells
Hooikaas, P. J., Damstra, H. G. J., Gros, O., van Riel, W. E., Martin, M., Smits, Y. T., van Loosdregt, J., Kapitein, L. C., Berger, F., & Akhmanova, A. S. (2020) · eLife · doi:10.7554/elife.62876 · Cited by 53 · see citing works
“Kinesin-4 family members are well known for their ability to limit MT growth in a variety of cellular processes (Bieling et al., 2010; Bringmann et al., 2004; He et al., 2014; Yue et al., 2018).”
BackgroundRegulation of ciliary membrane protein trafficking and signalling by kinesin motor proteins
Morthorst, S. K., Christensen, S. T., & Pedersen, L. B. (2018) · The FEBS Journal · doi:10.1111/febs.14583 · Cited by 49 · see citing works
Intraflagellar transport is deeply integrated in hedgehog signaling
Eguether, T., Cordelières, F. P., & Pazour, G. J. (2018) · Molecular Biology of the Cell · doi:10.1091/mbc.e17-10-0600 · Cited by 52 · see citing works
“Like Gli2, Kif7 and SuFu also localize to the ciliary tip, and the amount at the tip increases on pathway activation (Haycraft et al., 2005; He et al., 2014).”
BackgroundThe hedgehog pathway in nonalcoholic fatty liver disease
Machado, M. V., & Diehl, A. M. (2018) · Critical Reviews in Biochemistry and Molecular Biology · doi:10.1080/10409238.2018.1448752 · Cited by 52 · see citing works
“Kif7, which also localizes to the tip of the PC when Smo is active, enhances Gli–Sufu localization in this compartment (He et al. 2014), and full-length Gli dissociates from Sufu, moves back to the cytoplasm, and enters the nucleus, where it promotes the transcription of several genes, including…”
BackgroundA CEP104-CSPP1 Complex Is Required for Formation of Primary Cilia Competent in Hedgehog Signaling
Frikstad, K.-A. M., Molinari, E., Thoresen, M., Ramsbottom, S. A., Hughes, F. E. P., Letteboer, S. J. F., Gilani, S., Schink, K. O., Stokke, T., Geimer, S., et al. (2019) · Cell Reports · doi:10.1016/j.celrep.2019.07.025 · Cited by 49 · see citing works
“Speculatively, the CSPP-L/CEP104 complex hence opposes KIF7 and KIAA0556mediated axoneme growth restriction at the ciliary tip (He et al., 2014; Sanders et al., 2015).”
BackgroundInfluentialKanamaru, T., Neuner, A., Kurtulmus, B., & Pereira, G. (2021) · The EMBO Journal · doi:10.15252/embj.2021108843 · Cited by 43 · see citing works
Hedgehog signaling: From basic research to clinical applications
Yao, E., & Chuang, P. (2015) · Journal of the Formosan Medical Association · doi:10.1016/j.jfma.2015.01.005 · Cited by 48 · see citing works
“Kif7 controls cilium architecture at the tip and this could affect Sufu-Gli activity.(78) New tools are needed in order to establish the function of ciliary localization of Hh pathway components.”
BackgroundThe heterotrimeric kinesin-2 complex interacts with and regulates GLI protein function
Carpenter, B. S., Barry, R. L., Verhey, K. J., & Allen, B. L. (2015) · Journal of Cell Science · doi:10.1242/jcs.162552 · Cited by 48 · see citing works
“However, given that KIF7 does not possess plus-end-directed microtubule motor activity (He et al., 2014), one possibility is that KAP3–GLI protein interactions are disrupted in Kif7-mutant cells owing to the disorganized ciliary tip compartments.”
BackgroundResultInfluentialHan, Y., Xiong, Y., Shi, X., Wu, J. I., Zhao, Y., & Jiang, J. (2017) · PLoS Biology · doi:10.1371/journal.pbio.2002063 · Cited by 48 · see citing works
Cell Cycle-Related Kinase (CCRK) regulates ciliogenesis and Hedgehog signaling in mice
Snouffer, A., Brown, D. A., Lee, H., Walsh, J. D., Lupu, F. I., Norman, R. X., Lechtreck, K. F., Ko, H. W., & Eggenschwiler, J. T. (2017) · PLoS Genetics · doi:10.1371/journal.pgen.1006912 · Cited by 48 · see citing works
“Although loss of the ciliary kinesin Kif7 and loss of CCRK each cause deregulation of ciliary length, their effects on Hh pathway activity and neural patterning are largely opposite from one another [43, 44].”
Wormser, O., Gradstein, L., Yogev, Y., Perez, Y., Kadir, R., Goliand, I., Sadaka, Y., Riati, S. E., Flusser, H., Nachmias, D., et al. (2019) · European Journal of Human Genetics · doi:10.1038/s41431-019-0347-z · Cited by 48 · see citing works
Goetz, S. C., Bangs, F. K., Barrington, C. L., Katsanis, N., & Anderson, K. V. (2017) · PLoS ONE · doi:10.1371/journal.pone.0173399 · Cited by 45 · see citing works
Self-citationTruncated SALL1 Impedes Primary Cilia Function in Townes-Brocks Syndrome
Bozal‐Basterra, L., Martín‐Ruiz, I., Pirone, L., Liang, Y., Sigurðsson, J. O., Gonzalez-Santamarta, M., Giordano, I., Gabicagogeascoa, E., de Luca, A., Rodríguez, J. A., et al. (2018) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2017.12.017 · Cited by 42 · see citing works
Self-citationInvolvement of Wnt signaling in primary cilia assembly and disassembly
Lee, K. H. (2020) · FEBS Journal · doi:10.1111/febs.15579 · Cited by 40 · see citing works
Ververis, A., Christodoulou, A., Christoforou, M., Kamilari, C., Lederer, C. W., & Santama, N. (2015) · Cellular and Molecular Life Sciences · doi:10.1007/s00018-015-1980-5 · Cited by 41 · see citing works
Causes, costs and consequences of kinesin motors communicating through the microtubule lattice
Verhey, K. J., & Ohi, R. (2023) · Journal of Cell Science · doi:10.1242/jcs.260735 · Cited by 37 · see citing works
Ritter, A., Kreis, N., Roth, S., Friemel, A., Jennewein, L., Eichbaum, C., Solbach, C., Louwen, F., & Yuan, J. (2019) · Stem Cell Research & Therapy · doi:10.1186/s13287-019-1373-z · Cited by 40 · see citing works
- Authors
- He, M., Subramanian, R., Bangs, F., Omelchenko, T., Liem Jr, K. F., Kapoor, T. M., & Anderson, K. V.
- Affiliations
- Memorial Sloan Kettering Cancer Center (US)
- Rockefeller University (US)
- Corresponding author
- Anderson, Kathryn V.
- Venue
- Nature Cell Biology
- Published
- 2014-06-22
- Volume
- 16
- Issue
- 7
- Pages
- 663-672
- ISSN
- 1465-7392 (ISSN-L)
- 1476-4679
- PMC
- PMC4085576
- OpenAlex
- W2083616180
- Cited by
- 302
- References
- 55
- Citations by year
- 2026: 14 · 2025: 20 · 2024: 20 · 2023: 13 · 2022: 30 · 2021: 26 · 2020: 26 · 2019: 27 · 2018: 31 · 2017: 34 · 2016: 22 · 2015: 32 · 2014: 7
- FWCI
- 20.74
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Licence
- www.springer.com/tdm (tdm)
- Locations
- Nature Cell Biology (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (acceptedVersion)
- Funders
- National Cancer Institute (P30CA008748)
- National Institute of General Medical Sciences (R01GM065933, GM65933)
- National Institute of Neurological Disorders and Stroke (NS044385, R01NS044385)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Kidney Cyst Diseases
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Hedgehog Signaling Pathway Studies
- Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Cystic Fibrosis Research Advances
- Keywords
- KIF7, kinesin-4, Hedgehog signaling, primary cilia, microtubule plus ends, ciliopathies, intraflagellar transport, ciliary trafficking
- MeSH terms
- Animals, Cell Line, Cells, Cultured, Cilia, Fibroblasts, Humans, Microtubules, Mutation, Protein Binding, Recombinant Proteins, Signal Transduction, Kinesins, Protein Transport, NIH 3T3 Cells, Mice, Hedgehog Proteins, Axoneme, HEK293 Cells
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
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