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References
Bangs, F., and Anderson, K.V. (2017). Primary cilia and mammalian hedgehog signaling. Cold Spring Harb Perspect Biol 9, a028175.
Bharti, P., Schliebs, W., Schievelbusch, T., Neuhaus, A., David, C., Kock, K., Herrmann, C., Meyer, H.E., Wiese, S., Warscheid, B., et al. (2011). PEX14 is required for microtubule-based peroxisome motility in human cells. J Cell Sci 124, 1759–1768.
Canterini, S., Dragotto, J., Dardis, A., Zampieri, S., De Stefano, M.E., Mangia, F., Erickson, R.P., and Fiorenza, M.T. (2017). Shortened primary cilium length and dysregulated Sonic hedgehog signaling in Niemann-Pick C1 disease. Hum Mol Genet 26, 2277–2289.
Chu, B.B., Liao, Y.C., Qi, W., Xie, C., Du, X., Wang, J., Yang, H., Miao, H. H., Li, B.L., and Song, B.L. (2015). Cholesterol transport through lysosome-peroxisome membrane contacts. Cell 161, 291–306.
Das, A., Brown, M.S., Anderson, D.D., Goldstein, J.L., and Radhakrishnan, A. (2014). Three pools of plasma membrane cholesterol and their relation to cholesterol homeostasis. eLife 3, e02882.
Formichi, P., Battisti, C., De Santi, M.M., Guazzo, R., Tripodi, S.A., Radi, E., Rossi, B., Tarquini, E., and Federico, A. (2018). Primary cilium alterations and expression changes of Patched1 proteins in niemann-pick type C disease. J Cell Physiol 233, 663–672.
Gong, X., Qian, H., Cao, P., Zhao, X., Zhou, Q., Lei, J., and Yan, N. (2018). Structural basis for the recognition of Sonic Hedgehog by human Patched1. Science 361, eaas8935.
Hu, A., and Song, B.L. (2019). The interplay of Patched, Smoothened and cholesterol in Hedgehog signaling. Curr Opin Cell Biol 61, 31–38.
Kanerva, K., Uronen, R.L., Blom, T., Li, S., Bittman, R., Lappalainen, P., Peränen, J., Raposo, G., and Ikonen, E. (2013). LDL cholesterol recycles to the plasma membrane via a Rab8a-Myosin5b-actin-dependent membrane transport route. Dev Cell 27, 249–262.
Luo, J., Jiang, L., Yang, H., and Song, B.L. (2017). Routes and mechanisms of post-endosomal cholesterol trafficking: A story that never ends. Traffic 18, 209–217.
Luo, J., Jiang, L.Y., Yang, H., and Song, B.L. (2019). Intracellular cholesterol transport by sterol transfer proteins at membrane contact sites. Trends Biochem Sci 44, 273–292.
Maharjan, Y., Lee, J.N., Kwak, S.A., Dutta, R.K., Park, C., Choe, S.K., and Park, R. (2020). TMEM135 regulates primary ciliogenesis through modulation of intracellular cholesterol distribution. EMBO Rep 21, e48901.
Miyamoto, T., Hosoba, K., Itabashi, T., Iwane, A.H., Akutsu, S.N., Ochiai, H., Saito, Y., Yamamoto, T., and Matsuura, S. (2020). Insufficiency of ciliary cholesterol in hereditary Zellweger syndrome. EMBO J 39, e103499.
Neuhaus, A., Eggeling, C., Erdmann, R., and Schliebs, W. (2016). Why do peroxisomes associate with the cytoskeleton? Biochim Biophys Acta 1863, 1019–1026.
Porter, J.A., Young, K.E., and Beachy, P.A. (1996). Cholesterol modification of hedgehog signaling proteins in animal development. Science 274, 255–259.
Raleigh, D.R., Sever, N., Choksi, P.K., Sigg, M.A., Hines, K.M., Thompson, B.M., Elnatan, D., Jaishankar, P., Bisignano, P., Garcia-Gonzalo, F.R., et al. (2018). Cilia-associated oxysterols activate smoothened. Mol Cell 72, 316–327.e5.
Saheki, Y., and De Camilli, P. (2017). Endoplasmic reticulum-plasma membrane contact sites. Annu Rev Biochem 86, 659–684.
Sargsyan, Y., and Thoms, S. (2020). Staying in healthy contact: how peroxisomes interact with other cell organelles. Trends Mol Med 26, 201–214.
Soccio, R.E., and Breslow, J.L. (2004). Intracellular cholesterol transport. Arterioscler Thromb Vasc Biol 24, 1150–1160.
Suzuki, A., Ogata, K., Yoshioka, H., Shim, J., Wassif, C.A., Porter, F.D., and Iwata, J. (2020). Disruption of Dhcr7 and Insig1/2 in cholesterol metabolism causes defects in bone formation and homeostasis through primary cilium formation. Bone Res 8, 1.
Trinh, M.N., Brown, M.S., Goldstein, J.L., Han, J., Vale, G., McDonald, J. G., Seemann, J., Mendell, J.T., and Lu, F. (2020). Last step in the path of LDL cholesterol from lysosome to plasma membrane to ER is governed by phosphatidylserine. Proc Natl Acad Sci USA 117, 18521–18529.
Valm, A.M., Cohen, S., Legant, W.R., Melunis, J., Hershberg, U., Wait, E., Cohen, A.R., Davidson, M.W., Betzig, E., and Lippincott-Schwartz, J. (2017). Applying systems-level spectral imaging and analysis to reveal the organelle interactome. Nature 546, 162–167.
Wang, H., Ma, Q., Qi, Y., Dong, J., Du, X., Rae, J., Wang, J., Wu, W.F., Brown, A.J., Parton, R.G., et al. (2019). ORP2 delivers cholesterol to the plasma membrane in exchange for phosphatidylinositol 4,5-bisphosphate (PI(4,5)P2). Mol Cell 73, 458–473.e7.
Xiao, J., Luo, J., Hu, A., Xiao, T., Li, M., Kong, Z., Jiang, L., Zhou, Z., Liao, Y., Xie, C., et al. (2019). Cholesterol transport through the peroxisome-ER membrane contacts tethered by PI(4,5)P2 and extended synaptotagmins. Sci China Life Sci 62, 1117–1135.
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This work was supported by the National Natural Science Foundation of China (31771568, 32021003 and 91954203), and the Ministry of Science and Technology of China (2016YFA0500100).
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Luo, J., Liu, YB. & Song, BL. Hitching a ride to the top: peroxisomes fuel cilium with cholesterol. Sci. China Life Sci. 64, 478–481 (2021). https://doi.org/10.1007/s11427-020-1866-8
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DOI: https://doi.org/10.1007/s11427-020-1866-8