{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,18]],"date-time":"2026-10-18T07:09:49Z","timestamp":1792307389823,"version":"4.4.0"},"reference-count":87,"publisher":"Elsevier BV","issue":"3","license":[{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2025,3,1]],"date-time":"2025-03-01T00:00:00Z","timestamp":1740787200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2025,2,11]],"date-time":"2025-02-11T00:00:00Z","timestamp":1739232000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000738","name":"US Department of Veterans Affairs","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000738","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005188","name":"Tobacco-Related Disease Research Program","doi-asserted-by":"publisher","award":["T30FT1019"],"award-info":[{"award-number":["T30FT1019"]}],"id":[{"id":"10.13039\/100005188","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100014989","name":"The Chan Zuckerberg Initiative","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014989","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["1R01GM114414"],"award-info":[{"award-number":["1R01GM114414"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["R01NS120287"],"award-info":[{"award-number":["R01NS120287"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000879","name":"Alfred P. Sloan Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000879","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000011","name":"Howard Hughes Medical Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000011","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100020420","name":"Nan Fung Life Sciences","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100020420","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100019193","name":"Philomathia Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100019193","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006978","name":"University of California Berkeley","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006978","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["cell.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Cell Reports"],"published-print":{"date-parts":[[2025,3]]},"DOI":"10.1016\/j.celrep.2025.115383","type":"journal-article","created":{"date-parts":[[2025,3,8]],"date-time":"2025-03-08T14:31:39Z","timestamp":1741444299000},"page":"115383","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":14,"title":["Multicilia dynamically transduce Sonic Hedgehog signaling to regulate choroid plexus functions"],"prefix":"10.1016","volume":"44","author":[{"given":"Suifang","family":"Mao","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rui","family":"Song","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shibo","family":"Jin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Song","family":"Pang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aleksandra","family":"Jovanovic","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adam","family":"Zimmerman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peng","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xinying","family":"Wu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael F.","family":"Wendland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kerry","family":"Lin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wei-Chi","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Semil P.","family":"Choksi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gang","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael J.","family":"Holtzman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeremy F.","family":"Reiter","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Wan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhenyu","family":"Xuan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yang K.","family":"Xiang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C. Shan","family":"Xu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Srigokul","family":"Upadhyayula","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harald F.","family":"Hess","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lin","family":"He","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.celrep.2025.115383_bib1","doi-asserted-by":"crossref","first-page":"17553","DOI":"10.1523\/JNEUROSCI.3258-13.2013","article-title":"The Choroid Plexus and Cerebrospinal Fluid: Emerging Roles in Development, Disease, and Therapy","volume":"33","author":"Lehtinen","year":"2013","journal-title":"J. Neurosci."},{"key":"10.1016\/j.celrep.2025.115383_bib2","doi-asserted-by":"crossref","first-page":"445","DOI":"10.1038\/nrn3921","article-title":"Development and functions of the choroid plexus\u2013cerebrospinal fluid system","volume":"16","author":"Lun","year":"2015","journal-title":"Nat. Rev. Neurosci."},{"key":"10.1016\/j.celrep.2025.115383_bib3","doi-asserted-by":"crossref","DOI":"10.1016\/j.celrep.2024.114331","article-title":"The evolutionarily conserved choroid plexus contributes to the homeostasis of brain ventricles in zebrafish","volume":"43","author":"Jeong","year":"2024","journal-title":"Cell Rep."},{"key":"10.1016\/j.celrep.2025.115383_bib4","doi-asserted-by":"crossref","first-page":"261","DOI":"10.1016\/j.devcel.2020.01.027","article-title":"Emergence and Developmental Roles of the Cerebrospinal Fluid System","volume":"52","author":"Fame","year":"2020","journal-title":"Dev. Cell"},{"key":"10.1016\/j.celrep.2025.115383_bib5","doi-asserted-by":"crossref","first-page":"76","DOI":"10.1096\/fj.04-1711fje","article-title":"Reduced cerebrospinal fluid production and intracranial pressure in mice lacking choroid plexus water channel Aquaporin-1","volume":"19","author":"Oshio","year":"2005","journal-title":"FASEB J."},{"key":"10.1016\/j.celrep.2025.115383_bib6","doi-asserted-by":"crossref","first-page":"62","DOI":"10.1186\/s12987-022-00360-w","article-title":"Posthemorrhagic hydrocephalus associates with elevated inflammation and CSF hypersecretion via activation of choroidal transporters","volume":"19","author":"Lolansen","year":"2022","journal-title":"Fluids Barriers CNS"},{"key":"10.1016\/j.celrep.2025.115383_bib7","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1016\/0006-8993(73)90668-9","article-title":"Postnatal development of bulk flow in the cerebrospinal fluid system of the albino rat: Clearance of carboxyl-[14C]inulin after intrathecal infusion","volume":"52","author":"Bass","year":"1973","journal-title":"Brain Res."},{"key":"10.1016\/j.celrep.2025.115383_bib8","doi-asserted-by":"crossref","first-page":"851","DOI":"10.1002\/dneu.22622","article-title":"Cerebrospinal fluid flow increases from newborn to adult stages","volume":"78","author":"Di Palma","year":"2018","journal-title":"Dev. Neurobiol."},{"key":"10.1016\/j.celrep.2025.115383_bib9","doi-asserted-by":"crossref","first-page":"335","DOI":"10.1093\/cercor\/11.4.335","article-title":"Age-related Volumetric Changes of Brain Gray and White Matter in Healthy Infants and Children","volume":"11","author":"Matsuzawa","year":"2001","journal-title":"Cereb. Cortex"},{"key":"10.1016\/j.celrep.2025.115383_bib10","doi-asserted-by":"crossref","first-page":"1847","DOI":"10.1152\/physrev.00004.2013","article-title":"Cerebrospinal Fluid Secretion by the Choroid Plexus","volume":"93","author":"Damkier","year":"2013","journal-title":"Physiol. Rev."},{"key":"10.1016\/j.celrep.2025.115383_bib11","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1186\/s12987-020-00196-2","article-title":"Choroid plexus and the blood\u2013cerebrospinal fluid barrier in disease","volume":"17","author":"Sol\u00e1r","year":"2020","journal-title":"Fluids Barriers CNS"},{"key":"10.1016\/j.celrep.2025.115383_bib12","doi-asserted-by":"crossref","first-page":"S4","DOI":"10.1016\/j.diff.2011.11.002","article-title":"Structure and function of vertebrate cilia, towards a new taxonomy","volume":"83","author":"Takeda","year":"2012","journal-title":"Differentiation."},{"key":"10.1016\/j.celrep.2025.115383_bib13","doi-asserted-by":"crossref","first-page":"533","DOI":"10.1038\/nrm.2017.60","article-title":"Genes and molecular pathways underpinning ciliopathies","volume":"18","author":"Reiter","year":"2017","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib14","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1038\/s41581-019-0116-9","article-title":"Cellular signalling by primary cilia in development, organ function and disease","volume":"15","author":"Anvarian","year":"2019","journal-title":"Nat. Rev. Nephrol."},{"key":"10.1016\/j.celrep.2025.115383_bib15","doi-asserted-by":"crossref","first-page":"815","DOI":"10.1242\/bio.20121081","article-title":"Proteomic analysis of multiple primary cilia reveals a novel mode of ciliary development in mammals","volume":"1","author":"Narita","year":"2012","journal-title":"Biol. Open"},{"key":"10.1016\/j.celrep.2025.115383_bib16","doi-asserted-by":"crossref","first-page":"629","DOI":"10.1126\/science.1124534","article-title":"The Primary Cilium as the Cell\u2019s Antenna: Signaling at a Sensory Organelle","volume":"313","author":"Singla","year":"2006","journal-title":"Science"},{"key":"10.1016\/j.celrep.2025.115383_bib17","doi-asserted-by":"crossref","first-page":"32","DOI":"10.1016\/j.cell.2009.03.023","article-title":"The Vertebrate Primary Cilium in Development, Homeostasis, and Disease","volume":"137","author":"Gerdes","year":"2009","journal-title":"Cell"},{"key":"10.1016\/j.celrep.2025.115383_bib18","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.53580","article-title":"Super-resolution microscopy reveals coupling between mammalian centriole subdistal appendages and distal appendages","volume":"9","author":"Chong","year":"2020","journal-title":"Elife"},{"key":"10.1016\/j.celrep.2025.115383_bib19","doi-asserted-by":"crossref","first-page":"209","DOI":"10.1016\/j.devcel.2020.09.015","article-title":"Comparative Super-Resolution Mapping of Basal Feet Reveals a Modular but Distinct Architecture in Primary and Motile Cilia","volume":"55","author":"Nguyen","year":"2020","journal-title":"Dev. Cell"},{"key":"10.1016\/j.celrep.2025.115383_bib20","doi-asserted-by":"crossref","first-page":"R973","DOI":"10.1016\/j.cub.2014.08.047","article-title":"Multiciliated Cells","volume":"24","author":"Brooks","year":"2014","journal-title":"Curr. Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib21","doi-asserted-by":"crossref","first-page":"423","DOI":"10.1038\/nrm.2017.21","article-title":"The development and functions of multiciliated epithelia","volume":"18","author":"Spassky","year":"2017","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib22","doi-asserted-by":"crossref","first-page":"2641","DOI":"10.1016\/j.devcel.2023.10.003","article-title":"Choroid plexuses carry nodal-like cilia that undergo axoneme regression from early adult stage","volume":"58","author":"Ho","year":"2023","journal-title":"Dev. Cell"},{"key":"10.1016\/j.celrep.2025.115383_bib23","doi-asserted-by":"crossref","first-page":"797","DOI":"10.1002\/cm.21132","article-title":"Developmental changes in ciliary motility on choroid plexus epithelial cells during the perinatal period","volume":"70","author":"Nonami","year":"2013","journal-title":"Cytoskeleton"},{"key":"10.1016\/j.celrep.2025.115383_bib24","doi-asserted-by":"crossref","first-page":"4021","DOI":"10.1242\/dev.041129","article-title":"FoxJ1-dependent gene expression is required for differentiation of radial glia into ependymal cells and a subset of astrocytes in the postnatal brain","volume":"136","author":"Jacquet","year":"2009","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib25","doi-asserted-by":"crossref","DOI":"10.1242\/dev.154500","article-title":"A mutation in Ccdc39 causes neonatal hydrocephalus with abnormal motile cilia development in mice","volume":"145","author":"Abdelhamed","year":"2018","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib26","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1186\/s12987-021-00265-0","article-title":"The regulatory roles of motile cilia in CSF circulation and hydrocephalus","volume":"18","author":"Kumar","year":"2021","journal-title":"Fluids Barriers CNS"},{"key":"10.1016\/j.celrep.2025.115383_bib27","doi-asserted-by":"crossref","DOI":"10.3389\/fnmol.2022.927479","article-title":"Ependymal Cilia: Physiology and Role in Hydrocephalus","volume":"15","author":"Ji","year":"2022","journal-title":"Front. Mol. Neurosci."},{"key":"10.1016\/j.celrep.2025.115383_bib28","doi-asserted-by":"crossref","first-page":"3094","DOI":"10.1073\/pnas.94.7.3094","article-title":"The winged helix transcription factor HFH-4 is expressed during choroid plexus epithelial development in the mouse embryo","volume":"94","author":"Lim","year":"1997","journal-title":"Proc. Natl. Acad. Sci."},{"key":"10.1016\/j.celrep.2025.115383_bib29","doi-asserted-by":"crossref","first-page":"350","DOI":"10.1002\/dvg.22753","article-title":"A Knock-in Foxj1 CreERT2::GFP mouse for recombination in epithelial cells with motile cilia","volume":"52","author":"Muthusamy","year":"2014","journal-title":"genesis"},{"key":"10.1016\/j.celrep.2025.115383_bib30","doi-asserted-by":"crossref","first-page":"1077","DOI":"10.1172\/JCI4786","article-title":"Mutation of the mouse hepatocyte nuclear factor\/forkhead homologue 4 gene results in an absence of cilia and random left-right asymmetry","volume":"102","author":"Chen","year":"1998","journal-title":"J. Clin. Investig."},{"key":"10.1016\/j.celrep.2025.115383_bib31","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1165\/ajrcmb.23.1.4070","article-title":"Ciliogenesis and Left\u2013Right Axis Defects in Forkhead Factor HFH-4\u2013Null Mice","volume":"23","author":"Brody","year":"2000","journal-title":"Am. J. Respir. Cell Mol. Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib32","doi-asserted-by":"crossref","first-page":"12","DOI":"10.1186\/2046-2530-2-12","article-title":"Ciliogenesis and cerebrospinal fluid flow in the developing Xenopus brain are regulated by foxj1","volume":"2","author":"Hagenlocher","year":"2013","journal-title":"Cilia"},{"key":"10.1016\/j.celrep.2025.115383_bib33","doi-asserted-by":"crossref","DOI":"10.1016\/j.celrep.2021.109775","article-title":"Diversity and function of motile ciliated cell types within ependymal lineages of the zebrafish brain","volume":"37","author":"D\u2019Gama","year":"2021","journal-title":"Cell Rep."},{"key":"10.1016\/j.celrep.2025.115383_bib34","doi-asserted-by":"crossref","first-page":"3037","DOI":"10.1242\/dev.050120","article-title":"Planar polarity of multiciliated ependymal cells involves the anterior migration of basal bodies regulated by non-muscle myosin II","volume":"137","author":"Hirota","year":"2010","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib35","doi-asserted-by":"crossref","first-page":"543","DOI":"10.1016\/j.tins.2016.05.004","article-title":"Planar Organization of Multiciliated Ependymal (E1) Cells in the Brain Ventricular Epithelium","volume":"39","author":"Ohata","year":"2016","journal-title":"Trends Neurosci."},{"key":"10.1016\/j.celrep.2025.115383_bib36","doi-asserted-by":"crossref","first-page":"2279","DOI":"10.1038\/s41467-018-04676-w","article-title":"Ependymal cilia beating induces an actin network to protect centrioles against shear stress","volume":"9","author":"Mahuzier","year":"2018","journal-title":"Nat. Commun."},{"key":"10.1016\/j.celrep.2025.115383_bib37","doi-asserted-by":"crossref","first-page":"1276","DOI":"10.1242\/dev.072728","article-title":"Differential regulation of node formation, nodal ciliogenesis and cilia positioning by Noto and Foxj1","volume":"139","author":"Alten","year":"2012","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib38","doi-asserted-by":"crossref","first-page":"1427","DOI":"10.1242\/dev.074666","article-title":"Switching on cilia: transcriptional networks regulating ciliogenesis","volume":"141","author":"Choksi","year":"2014","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib39","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pbio.3002468","article-title":"The forkhead transcription factor Foxj1 controls vertebrate olfactory cilia biogenesis and sensory neuron differentiation","volume":"22","author":"Rayamajhi","year":"2024","journal-title":"PLoS Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib40","doi-asserted-by":"crossref","first-page":"341","DOI":"10.1038\/ncb2040","article-title":"Coupling between hydrodynamic forces and planar cell polarity orients mammalian motile cilia","volume":"12","author":"Guirao","year":"2010","journal-title":"Nat. Cell Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib41","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1523\/JNEUROSCI.1108-04.2005","article-title":"Adult Ependymal Cells Are Postmitotic and Are Derived from Radial Glial Cells during Embryogenesis","volume":"25","author":"Spassky","year":"2005","journal-title":"J. Neurosci."},{"key":"10.1016\/j.celrep.2025.115383_bib42","doi-asserted-by":"crossref","first-page":"2600","DOI":"10.1523\/JNEUROSCI.3744-09.2010","article-title":"Cilia Organize Ependymal Planar Polarity","volume":"30","author":"Mirzadeh","year":"2010","journal-title":"J. Neurosci."},{"key":"10.1016\/j.celrep.2025.115383_bib43","doi-asserted-by":"crossref","first-page":"558","DOI":"10.1016\/j.neuron.2014.06.022","article-title":"Loss of Dishevelleds Disrupts Planar Polarity in Ependymal Motile Cilia and Results in Hydrocephalus","volume":"83","author":"Ohata","year":"2014","journal-title":"Neuron"},{"key":"10.1016\/j.celrep.2025.115383_bib44","doi-asserted-by":"crossref","first-page":"1695","DOI":"10.1016\/j.addr.2004.07.005","article-title":"The structure of the choroid plexus and the physiology of the choroid plexus epithelium","volume":"56","author":"Redzic","year":"2004","journal-title":"Adv. Drug Deliv. Rev."},{"key":"10.1016\/j.celrep.2025.115383_bib45","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1113\/jphysiol.1970.sp009159","article-title":"The effects of some inhibitors and accelerators of sodium transport on the turnover of 22 Na in the cerebrospinal fluid and the brain","volume":"209","author":"Davson","year":"1970","journal-title":"J. Physiol."},{"key":"10.1016\/j.celrep.2025.115383_bib46","doi-asserted-by":"crossref","first-page":"5329","DOI":"10.1242\/dev.02153","article-title":"Dysfunctional cilia lead to altered ependyma and choroid plexus function,and result in the formation of hydrocephalus","volume":"132","author":"Banizs","year":"2005","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib47","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1038\/nature02061","article-title":"Hedgehog signalling in the mouse requires intraflagellar transport proteins","volume":"426","author":"Huangfu","year":"2003","journal-title":"Nature"},{"key":"10.1016\/j.celrep.2025.115383_bib48","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1038\/nrg2774","article-title":"The primary cilium: a signalling centre during vertebrate development","volume":"11","author":"Goetz","year":"2010","journal-title":"Nat. Rev. Genet."},{"key":"10.1016\/j.celrep.2025.115383_bib49","doi-asserted-by":"crossref","DOI":"10.1101\/cshperspect.a028175","article-title":"Primary Cilia and Mammalian Hedgehog Signaling","volume":"9","author":"Bangs","year":"2017","journal-title":"Cold Spring Harb. Perspect. Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib50","doi-asserted-by":"crossref","first-page":"372","DOI":"10.1126\/science.1139740","article-title":"Patched1 Regulates Hedgehog Signaling at the Primary Cilium","volume":"317","author":"Rohatgi","year":"2007","journal-title":"Science"},{"key":"10.1016\/j.celrep.2025.115383_bib51","doi-asserted-by":"crossref","first-page":"1018","DOI":"10.1038\/nature04117","article-title":"Vertebrate Smoothened functions at the primary cilium","volume":"437","author":"Corbit","year":"2005","journal-title":"Nature"},{"key":"10.1016\/j.celrep.2025.115383_bib52","doi-asserted-by":"crossref","first-page":"2535","DOI":"10.1242\/dev.033795","article-title":"Sonic hedgehog signaling regulates a novel epithelial progenitor domain of the hindbrain choroid plexus","volume":"136","author":"Huang","year":"2009","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib53","doi-asserted-by":"crossref","first-page":"62","DOI":"10.1016\/j.tips.2015.09.001","article-title":"Smoothened Regulation: A Tale of Two Signals","volume":"37","author":"Arensdorf","year":"2016","journal-title":"Trends Pharmacol. Sci."},{"key":"10.1016\/j.celrep.2025.115383_bib54","doi-asserted-by":"crossref","first-page":"11","DOI":"10.3390\/jdb5040011","article-title":"Contributions of Noncanonical Smoothened Signaling During Embryonic Development","volume":"5","author":"Pandit","year":"2017","journal-title":"J. Dev. Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib55","doi-asserted-by":"crossref","first-page":"312","DOI":"10.1016\/j.cell.2018.04.029","article-title":"Structural Basis of Smoothened Activation in Hedgehog Signaling","volume":"174","author":"Huang","year":"2018","journal-title":"Cell"},{"key":"10.1016\/j.celrep.2025.115383_bib56","doi-asserted-by":"crossref","first-page":"279","DOI":"10.1038\/s41586-019-1286-0","article-title":"Cryo-EM structure of oxysterol-bound human Smoothened coupled to a heterotrimeric Gi","volume":"571","author":"Qi","year":"2019","journal-title":"Nature"},{"key":"10.1016\/j.celrep.2025.115383_bib57","doi-asserted-by":"crossref","DOI":"10.1073\/pnas.2300919120","article-title":"Fully activated structure of the sterol-bound Smoothened GPCR-Gi protein complex","volume":"120","author":"Vo","year":"2023","journal-title":"Proc. Natl. Acad. Sci."},{"key":"10.1016\/j.celrep.2025.115383_bib58","doi-asserted-by":"crossref","first-page":"967","DOI":"10.1038\/nature07459","article-title":"G protein G\u03b1i functions immediately downstream of Smoothened in Hedgehog signalling","volume":"456","author":"Ogden","year":"2008","journal-title":"Nature"},{"key":"10.1016\/j.celrep.2025.115383_bib59","doi-asserted-by":"crossref","first-page":"C1409","DOI":"10.1152\/ajpcell.00408.2006","article-title":"Altered pH i regulation and Na +\/HCO 3 \u2212 transporter activity in choroid plexus of cilia-defective Tg737 orpk mutant mouse","volume":"292","author":"Banizs","year":"2007","journal-title":"Am. J. Physiol. Cell Physiol."},{"key":"10.1016\/j.celrep.2025.115383_bib60","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0122513","article-title":"Fourth-Generation Epac-Based FRET Sensors for cAMP Feature Exceptional Brightness, Photostability and Dynamic Range: Characterization of Dedicated Sensors for FLIM, for Ratiometry and with High Affinity","volume":"10","author":"Klarenbeek","year":"2015","journal-title":"PLoS One"},{"key":"10.1016\/j.celrep.2025.115383_bib61","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.25916","article-title":"Enhanced FIB-SEM systems for large-volume 3D imaging","volume":"6","author":"Xu","year":"2017","journal-title":"Elife"},{"key":"10.1016\/j.celrep.2025.115383_bib62","doi-asserted-by":"crossref","first-page":"147","DOI":"10.1038\/s41586-021-03992-4","article-title":"An open-access volume electron microscopy atlas of whole cells and tissues","volume":"599","author":"Xu","year":"2021","journal-title":"Nature"},{"key":"10.1016\/j.celrep.2025.115383_bib63","first-page":"777","article-title":"A morphological study of the development of the mouse choroid plexus","volume":"129","author":"Sturrock","year":"1979","journal-title":"J. Anat."},{"key":"10.1016\/j.celrep.2025.115383_bib64","doi-asserted-by":"crossref","first-page":"447","DOI":"10.1038\/s41467-020-20666-3","article-title":"Choroid plexus NKCC1 mediates cerebrospinal fluid clearance during mouse early postnatal development","volume":"12","author":"Xu","year":"2021","journal-title":"Nat. Commun."},{"key":"10.1016\/j.celrep.2025.115383_bib65","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0065629","article-title":"Mechanisms That Determine the Internal Environment of the Developing Brain: A Transcriptomic, Functional and Ultrastructural Approach","volume":"8","author":"Liddelow","year":"2013","journal-title":"PLoS One"},{"key":"10.1016\/j.celrep.2025.115383_bib66","doi-asserted-by":"crossref","first-page":"919","DOI":"10.1152\/physrev.00060.2021","article-title":"The choroid plexus: a missing link in our understanding of brain development and function","volume":"103","author":"Saunders","year":"2023","journal-title":"Physiol. Rev."},{"key":"10.1016\/j.celrep.2025.115383_bib67","doi-asserted-by":"crossref","first-page":"61","DOI":"10.1016\/S0092-8674(03)00511-7","article-title":"Two Populations of Node Monocilia Initiate Left-Right Asymmetry in the Mouse","volume":"114","author":"McGrath","year":"2003","journal-title":"Cell"},{"key":"10.1016\/j.celrep.2025.115383_bib68","doi-asserted-by":"crossref","DOI":"10.1126\/sciadv.abq8486","article-title":"Islet primary cilia motility controls insulin secretion","volume":"8","author":"Cho","year":"2022","journal-title":"Sci. Adv."},{"key":"10.1016\/j.celrep.2025.115383_bib69","doi-asserted-by":"crossref","first-page":"972","DOI":"10.1126\/science.abm1962","article-title":"Rhythmic cilia changes support SCN neuron coherence in circadian clock","volume":"380","author":"Tu","year":"2023","journal-title":"Science"},{"key":"10.1016\/j.celrep.2025.115383_bib70","doi-asserted-by":"crossref","first-page":"443","DOI":"10.1002\/ar.1091760407","article-title":"A comparative ultrastructural study of the choroid plexuses of the immature pig","volume":"176","author":"Davis","year":"1973","journal-title":"Anat. Rec."},{"key":"10.1016\/j.celrep.2025.115383_bib71","doi-asserted-by":"crossref","first-page":"325","DOI":"10.1002\/ar.1091940303","article-title":"The choroid plexus of the mature and aging rat: The choroidal epithelium","volume":"194","author":"Peters","year":"1979","journal-title":"Anat. Rec."},{"key":"10.1016\/j.celrep.2025.115383_bib72","series-title":"Epithelial Cell Culture Protocols Methods in Molecular Biology","first-page":"13","article-title":"Culture of Choroid Plexus Epithelial Cells and In Vitro Model of Blood\u2013CSF Barrier","author":"Monnot","year":"2012"},{"key":"10.1016\/j.celrep.2025.115383_bib73","doi-asserted-by":"crossref","first-page":"C1","DOI":"10.1152\/ajpcell.00374.2021","article-title":"Porcine choroid plexus-Riems cell line demonstrates altered polarization of transport proteins compared with the native epithelium","volume":"323","author":"Hochstetler","year":"2022","journal-title":"Am. J. Physiol. Cell Physiol."},{"key":"10.1016\/j.celrep.2025.115383_bib74","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1242\/dev.02732","article-title":"Intraflagellar transport is essential for endochondral bone formation","volume":"134","author":"Haycraft","year":"2007","journal-title":"Development"},{"key":"10.1016\/j.celrep.2025.115383_bib75","doi-asserted-by":"crossref","first-page":"515","DOI":"10.1165\/rcmb.2006-0475RC","article-title":"A Transgenic FOXJ1-Cre System for Gene Inactivation in Ciliated Epithelial Cells","volume":"36","author":"Zhang","year":"2007","journal-title":"Am. J. Respir. Cell Mol. Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib76","doi-asserted-by":"crossref","first-page":"418","DOI":"10.1038\/ncb3327","article-title":"Sonic Hedgehog promotes proliferation of Notch-dependent monociliated choroid plexus tumour cells","volume":"18","author":"Li","year":"2016","journal-title":"Nat. Cell Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib77","doi-asserted-by":"crossref","DOI":"10.3389\/fphys.2013.00344","article-title":"Polarization of membrane associated proteins in the choroid plexus epithelium from normal and slc4a10 knockout mice","volume":"4","author":"Christensen","year":"2013","journal-title":"Front. Physiol."},{"key":"10.1016\/j.celrep.2025.115383_bib78","doi-asserted-by":"crossref","first-page":"227","DOI":"10.1111\/j.1365-2818.2011.03526.x","article-title":"Freeze substitution in 3 h or less","volume":"243","author":"McDonald","year":"2011","journal-title":"J. Microsc."},{"key":"10.1016\/j.celrep.2025.115383_bib79","first-page":"2638","article-title":"Mammary collective cell migration involves transient loss of epithelial features and individual cell migration within the epithelium","volume":"125","author":"Ewald","year":"2012","journal-title":"J. Cell Sci."},{"key":"10.1016\/j.celrep.2025.115383_bib80","doi-asserted-by":"crossref","first-page":"269","DOI":"10.1016\/bs.mcb.2023.01.019","article-title":"Methods of enhanced FIB-SEM sample preparation and image acquisition","volume":"177","author":"Pang","year":"2023","journal-title":"Methods Cell Biol."},{"key":"10.1016\/j.celrep.2025.115383_bib81","doi-asserted-by":"crossref","first-page":"1226","DOI":"10.1038\/s41592-019-0582-9","article-title":"ilastik: interactive machine learning for (bio)image analysis","volume":"16","author":"Berg","year":"2019","journal-title":"Nat. Methods"},{"key":"10.1016\/j.celrep.2025.115383_bib82","doi-asserted-by":"crossref","first-page":"676","DOI":"10.1038\/nmeth.2019","article-title":"Fiji: an open-source platform for biological-image analysis","volume":"9","author":"Schindelin","year":"2012","journal-title":"Nat. Methods"},{"key":"10.1016\/j.celrep.2025.115383_bib83","doi-asserted-by":"crossref","first-page":"1116","DOI":"10.1016\/j.neuroimage.2006.01.015","article-title":"User-guided 3D active contour segmentation of anatomical structures: Significantly improved efficiency and reliability","volume":"31","author":"Yushkevich","year":"2006","journal-title":"Neuroimage"},{"key":"10.1016\/j.celrep.2025.115383_bib84","doi-asserted-by":"crossref","first-page":"1567","DOI":"10.1085\/jgp.201812119","article-title":"Illuminating cell signaling with genetically encoded FRET biosensors in adult mouse cardiomyocytes","volume":"150","author":"Reddy","year":"2018","journal-title":"J. Gen. Physiol."},{"key":"10.1016\/j.celrep.2025.115383_bib85","doi-asserted-by":"crossref","first-page":"13","DOI":"10.1186\/1743-8454-3-13","article-title":"Choroid plexus epithelial monolayers \u2013 a cell culture model from porcine brain","volume":"3","author":"Baehr","year":"2006","journal-title":"Cerebrospinal Fluid Res."},{"key":"10.1016\/j.celrep.2025.115383_bib86","doi-asserted-by":"crossref","first-page":"525","DOI":"10.1038\/nbt.3519","article-title":"Near-optimal probabilistic RNA-seq quantification","volume":"34","author":"Bray","year":"2016","journal-title":"Nat. Biotechnol."},{"key":"10.1016\/j.celrep.2025.115383_bib87","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1093\/nar\/gkn923","article-title":"Bioinformatics enrichment tools: paths toward the comprehensive functional analysis of large gene lists","volume":"37","author":"Huang","year":"2009","journal-title":"Nucleic Acids Res."}],"container-title":["Cell Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2211124725001548?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2211124725001548?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,4,23]],"date-time":"2026-04-23T04:49:56Z","timestamp":1776919796000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S2211124725001548"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3]]},"references-count":87,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2025,3]]}},"alternative-id":["S2211124725001548"],"URL":"https:\/\/doi.org\/10.1016\/j.celrep.2025.115383","relation":{},"ISSN":["2211-1247"],"issn-type":[{"value":"2211-1247","type":"print"}],"subject":[],"published":{"date-parts":[[2025,3]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Multicilia dynamically transduce Sonic Hedgehog signaling to regulate choroid plexus functions","name":"articletitle","label":"Article Title"},{"value":"Cell Reports","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.celrep.2025.115383","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2025 The Authors. Published by Elsevier Inc.","name":"copyright","label":"Copyright"}],"article-number":"115383"}}