About   Help   FAQ
Gene Expression Literature Summary
Symbol
Name
ID
Arl13b
ADP-ribosylation factor-like 13B
MGI:1915396

196 matching records from 196 references.

Summary by Age and Assay: Numbers in the table indicate the number of results matching the search criteria.
Age E7.5 E8 E8.5 E9 E9.5 E10 E10.5 E11.5 E12 E12.5 E13.5 E14 E14.5 E15.5 E16 E16.5 E17.5 E18.5 E P
Immunohistochemistry (section) 1 9 10 2 30 1 54 25 2 18 17 2 19 18 1 7 5 17 8 23
In situ RNA (section) 1
Immunohistochemistry (whole mount) 2 1 1 3 1 3 4 2 3 1 7
Western blot 3 1 1 1
RT-PCR 1 1 1 2

Summary by Gene and Reference: Number indicates the number of results matching the search criteria recorded for each reference.
* Indicates detailed expression data entries available
Arl13b  ADP-ribosylation factor-like 13B   (Synonyms: A530097K21Rik, A930014M17Rik, Arl2l1, C530009C10Rik, hnn)
Results  Reference
5J:314668 Abrams SR, Reiter JF, Ciliary Hedgehog signaling regulates cell survival to build the facial midline. Elife. 2021 Oct 21;10:e68558
1J:255486 Agbu SO, Liang Y, Liu A, Anderson KV, The small GTPase RSG1 controls a final step in primary cilia initiation. J Cell Biol. 2018 Jan 2;217(1):413-427
1J:333568 Akula SK, Marciano JH, Lim Y, Exposito-Alonso D, Hylton NK, Hwang GH, Neil JE, Dominado N, Bunton-Stasyshyn RK, Song JHT, Talukdar M, Schmid A, Teboul L, Mo A, Shin T, Finander B, Beck SG, Yeh RC, Otani A, Qian X, DeGennaro EM, Alkuraya FS, Maddirevula S, Cascino GD, Giannini C, Burrage LC, Rosenfield JA, Ketkar S, Clark GD, Bacino C, Lewis RA, Segal RA, Bazan JF, Smith KA, Golden JA, Cho G, Walsh CA, TMEM161B regulates cerebral cortical gyration, Sonic Hedgehog signaling, and ciliary structure in the developing central nervous system. Proc Natl Acad Sci U S A. 2023 Jan 24;120(4):e2209964120
2J:273860 Andreu-Cervera A, Anselme I, Karam A, Laclef C, Catala M, Schneider-Maunoury S, The Ciliopathy Gene Ftm/Rpgrip1l Controls Mouse Forebrain Patterning via Region-Specific Modulation of Hedgehog/Gli Signaling. J Neurosci. 2019 Mar 27;39(13):2398-2415
2*J:290235 Archambault D, Cheong A, Iverson E, Tremblay KD, Mager J, Protein phosphatase 1 regulatory subunit 35 is required for ciliogenesis, notochord morphogenesis, and cell-cycle progression during murine development. Dev Biol. 2020 Jul 3;465(1):1-10
1*J:290791 Baba Y, Watabe Y, Sagara H, Watanabe S, Sall1 plays pivotal roles for lens fiber cell differentiation in mouse. Biochem Biophys Res Commun. 2019 May 14;512(4):927-933
1J:346054 Baur K, Carrillo-Garcia C, San S, von Hahn M, Strelau J, Holzl-Wenig G, Mandl C, Ciccolini F, Growth/differentiation factor 15 controls ependymal and stem cell number in the V-SVZ. Stem Cell Reports. 2024 Mar 12;19(3):351-365
2J:347979 Baur K, San S, Holzl-Wenig G, Mandl C, Hellwig A, Ciccolini F, GDF15 controls primary cilia morphology and function thereby affecting progenitor proliferation. Life Sci Alliance. 2024 Jul;7(7)
1J:208632 Bazzi H, Anderson KV, Acentriolar mitosis activates a p53-dependent apoptosis pathway in the mouse embryo. Proc Natl Acad Sci U S A. 2014 Apr 15;111(15):E1491-500
1*J:178483 Berbari NF, Kin NW, Sharma N, Michaud EJ, Kesterson RA, Yoder BK, Mutations in Traf3ip1 reveal defects in ciliogenesis, embryonic development, and altered cell size regulation. Dev Biol. 2011 Dec 1;360(1):66-76
2J:171429 Besse L, Neti M, Anselme I, Gerhardt C, Ruther U, Laclef C, Schneider-Maunoury S, Primary cilia control telencephalic patterning and morphogenesis via Gli3 proteolytic processing. Development. 2011 May;138(10):2079-88
1*J:259558 Bhattacharyya S, Rainey MA, Arya P, Dutta S, George M, Storck MD, McComb RD, Muirhead D, Todd GL, Gould K, Datta K, Gelineau-van Waes J, Band V, Band H, Endocytic recycling protein EHD1 regulates primary cilia morphogenesis and SHH signaling during neural tube development. Sci Rep. 2016 Feb 17;6:20727
1J:182770 Bisgrove BW, Makova S, Yost HJ, Brueckner M, RFX2 is essential in the ciliated organ of asymmetry and an RFX2 transgene identifies a population of ciliated cells sufficient for fluid flow. Dev Biol. 2012 Mar 1;363(1):166-78
1*J:317479 Bizzotto S, Uzquiano A, Dingli F, Ershov D, Houllier A, Arras G, Richards M, Loew D, Minc N, Croquelois A, Houdusse A, Francis F, Eml1 loss impairs apical progenitor spindle length and soma shape in the developing cerebral cortex. Sci Rep. 2017 Dec 11;7(1):17308
1J:269515 Bowie E, Norris R, Anderson KV, Goetz SC, Spinocerebellar ataxia type 11-associated alleles of Ttbk2 dominantly interfere with ciliogenesis and cilium stability. PLoS Genet. 2018 Dec;14(12):e1007844
1J:266546 Brown JM, Garcia-Garcia MJ, Secretory pathway calcium ATPase 1 (SPCA1) controls mouse neural tube closure by regulating cytoskeletal dynamics. Development. 2018 Oct 5;145(19):dev170019
1J:158523 Brugmann SA, Allen NC, James AW, Mekonnen Z, Madan E, Helms JA, A primary cilia-dependent etiology for midline facial disorders. Hum Mol Genet. 2010 Apr 15;19(8):1577-92
3J:249800 Burnett JB, Lupu FI, Eggenschwiler JT, Proper ciliary assembly is critical for restricting Hedgehog signaling during early eye development in mice. Dev Biol. 2017 Oct 1;430(1):32-40
4J:301038 Casoni F, Croci L, Vincenti F, Podini P, Riba M, Massimino L, Cremona O, Consalez GG, ZFP423 regulates early patterning and multiciliogenesis in the hindbrain choroid plexus. Development. 2020 Nov 30;147(22):dev190173
4*J:120712 Caspary T, Larkins CE, Anderson KV, The graded response to sonic hedgehog depends on cilia architecture. Dev Cell. 2007 May;12(5):767-78
1*J:308674 Chang CF, Brown KM, Yang Y, Brugmann SA, Centriolar Protein C2cd3 Is Required for Craniofacial Development. Front Cell Dev Biol. 2021;9:647391
1J:236864 Chang CF, Chang YT, Millington G, Brugmann SA, Craniofacial Ciliopathies Reveal Specific Requirements for GLI Proteins during Development of the Facial Midline. PLoS Genet. 2016 Nov;12(11):e1006351
1J:222441 Chang R, Petersen JR, Niswander LA, Liu A, A hypomorphic allele reveals an important role of inturned in mouse skeletal development. Dev Dyn. 2015 Jun;244(6):736-47
2J:211665 Chaya T, Omori Y, Kuwahara R, Furukawa T, ICK is essential for cell type-specific ciliogenesis and the regulation of ciliary transport. EMBO J. 2014 Jun 2;33(11):1227-42
2J:218613 Chen J, Laclef C, Moncayo A, Snedecor ER, Yang N, Li L, Takemaru K, Paus R, Schneider-Maunoury S, Clark RA, The ciliopathy gene rpgrip1l is essential for hair follicle development. J Invest Dermatol. 2015 Mar;135(3):701-9
1J:288298 Chen KQ, Tahara N, Anderson A, Kawakami H, Kawakami S, Nishinakamura R, Pandolfi PP, Kawakami Y, Development of the Proximal-Anterior Skeletal Elements in the Mouse Hindlimb Is Regulated by a Transcriptional and Signaling Network Controlled by Sall4. Genetics. 2020 May;215(1):129-141
2*J:278736 Cheong A, Degani R, Tremblay KD, Mager J, A null allele of Dnaaf2 displays embryonic lethality and mimics human ciliary dyskinesia. Hum Mol Genet. 2019 Aug 15;28(16):2775-2784
1*J:178978 Chih B, Liu P, Chinn Y, Chalouni C, Komuves LG, Hass PE, Sandoval W, Peterson AS, A ciliopathy complex at the transition zone protects the cilia as a privileged membrane domain. Nat Cell Biol. 2011;14(1):61-72
1J:239874 Christ A, Christa A, Klippert J, Eule JC, Bachmann S, Wallace VA, Hammes A, Willnow TE, LRP2 Acts as SHH Clearance Receptor to Protect the Retinal Margin from Mitogenic Stimuli. Dev Cell. 2015 Oct 12;35(1):36-48
1J:181474 Christ A, Christa A, Kur E, Lioubinski O, Bachmann S, Willnow TE, Hammes A, LRP2 Is an Auxiliary SHH Receptor Required to Condition the Forebrain Ventral Midline for Inductive Signals. Dev Cell. 2012 Feb 14;22(2):268-78
1J:298672 Christ A, Marczenke M, Willnow TE, LRP2 controls sonic hedgehog-dependent differentiation of cardiac progenitor cells during outflow tract formation. Hum Mol Genet. 2020 Nov 25;29(19):3183-3196
1J:186552 Christopher KJ, Wang B, Kong Y, Weatherbee SD, Forward genetics uncovers Transmembrane protein 107 as a novel factor required for ciliogenesis and Sonic hedgehog signaling. Dev Biol. 2012 Aug 15;368(2):382-92
1J:309362 Cochrane CR, Vaghjiani V, Szczepny A, Jayasekara WSN, Gonzalez-Rajal A, Kikuchi K, McCaughan GW, Burgess A, Gough DJ, Watkins DN, Cain JE, Trp53 and Rb1 regulate autophagy and ligand-dependent Hedgehog signaling. J Clin Invest. 2020 Aug 3;130(8):4006-4018
1J:291480 Constable S, Long AB, Floyd KA, Schurmans S, Caspary T, The ciliary phosphatidylinositol phosphatase Inpp5e plays positive and negative regulatory roles in Shh signaling. Development. 2020 Feb 3;147(3):dev183301
1*J:226614 Costello I, Nowotschin S, Sun X, Mould AW, Hadjantonakis AK, Bikoff EK, Robertson EJ, Lhx1 functions together with Otx2, Foxa2, and Ldb1 to govern anterior mesendoderm, node, and midline development. Genes Dev. 2015 Oct 15;29(20):2108-22
2J:171221 Croyle MJ, Lehman JM, O'Connor AK, Wong SY, Malarkey EB, Iribarne D, Dowdle WE, Schoeb TR, Verney ZM, Athar M, Michaud EJ, Reiter JF, Yoder BK, Role of epidermal primary cilia in the homeostasis of skin and hair follicles. Development. 2011 May;138(9):1675-85
3J:166761 Cruz C, Ribes V, Kutejova E, Cayuso J, Lawson V, Norris D, Stevens J, Davey M, Blight K, Bangs F, Mynett A, Hirst E, Chung R, Balaskas N, Brody SL, Marti E, Briscoe J, Foxj1 regulates floor plate cilia architecture and modifies the response of cells to sonic hedgehog signalling. Development. 2010 Dec;137(24):4271-82
1J:309354 Cyge B, Voronina V, Hoque M, Kim EN, Hall J, Bailey-Lundberg JM, Pazour GJ, Crawford HC, Moon RT, Li FQ, Takemaru KI, Loss of the ciliary protein Chibby1 in mice leads to exocrine pancreatic degeneration and pancreatitis. Sci Rep. 2021 Aug 26;11(1):17220
2J:205544 Dai D, Li L, Huebner A, Zeng H, Guevara E, Claypool DJ, Liu A, Chen J, Planar cell polarity effector gene Intu regulates cell fate-specific differentiation of keratinocytes through the primary cilia. Cell Death Differ. 2013 Jan;20(1):130-8
1J:173143 Dai D, Zhu H, Wlodarczyk B, Zhang L, Li L, Li AG, Finnell RH, Roop DR, Chen J, Fuz controls the morphogenesis and differentiation of hair follicles through the formation of primary cilia. J Invest Dermatol. 2011 Feb;131(2):302-10
1J:306061 Damen M, Wirtz L, Soroka E, Khatif H, Kukat C, Simons BD, Bazzi H, High proliferation and delamination during skin epidermal stratification. Nat Commun. 2021 May 28;12(1):3227
2J:313289 Dias JM, Alekseenko Z, Jeggari A, Boareto M, Vollmer J, Kozhevnikova M, Wang H, Matise MP, Alexeyenko A, Iber D, Ericson J, A Shh/Gli-driven three-node timer motif controls temporal identity and fate of neural stem cells. Sci Adv. 2020 Sep;6(38)
1*J:284772 DiStasio A, Paulding D, Chaturvedi P, Stottmann RW, Nubp2 is required for cranial neural crest survival in the mouse. Dev Biol. 2020 Feb 15;458(2):189-199
1*J:178421 Dowdle WE, Robinson JF, Kneist A, Sirerol-Piquer MS, Frints SG, Corbit KC, Zaghloul NA, van Lijnschoten G, Mulders L, Verver DE, Zerres K, Reed RR, Attie-Bitach T, Johnson CA, Garcia-Verdugo JM, Katsanis N, Bergmann C, Reiter JF, Disruption of a ciliary B9 protein complex causes Meckel syndrome. Am J Hum Genet. 2011 Jul 15;89(1):94-110
3J:342023 Dubaic M, Peskova L, Hampl M, Weissova K, Celiker C, Shylo NA, Hruba E, Kavkova M, Zikmund T, Weatherbee SD, Kaiser J, Barta T, Buchtova M, Role of ciliopathy protein TMEM107 in eye development: insights from a mouse model and retinal organoid. Life Sci Alliance. 2023 Dec;6(12)
1J:354197 Dumoulin A, Wilson NH, Tucker KL, Stoeckli ET, A cell-autonomous role for primary cilium-mediated signaling in long-range commissural axon guidance. Development. 2024 Sep 1;151(17)
1J:335575 Duy PQ, Weise SC, Marini C, Li XJ, Liang D, Dahl PJ, Ma S, Spajic A, Dong W, Juusola J, Kiziltug E, Kundishora AJ, Koundal S, Pedram MZ, Torres-Fernandez LA, Handler K, De Domenico E, Becker M, Ulas T, Juranek SA, Cuevas E, Hao LT, Jux B, Sousa AMM, Liu F, Kim SK, Li M, Yang Y, Takeo Y, Duque A, Nelson-Williams C, Ha Y, Selvaganesan K, Robert SM, Singh AK, Allington G, Furey CG, Timberlake AT, Reeves BC, Smith H, Dunbar A, DeSpenza T Jr, Goto J, Marlier A, Moreno-De-Luca A, Yu X, Butler WE, Carter BS, Lake EMR, Constable RT, Rakic P, Lin H, Deniz E, Benveniste H, Malvankar NS, Estrada-Veras JI, Walsh CA, Alper SL, Schultze JL, Paeschke K, Doetzlhofer A, Wulczyn FG, Jin SC, Lifton RP, Sestan N, Kolanus W, Kahle KT, Impaired neurogenesis alters brain biomechanics in a neuroprogenitor-based genetic subtype of congenital hydrocephalus. Nat Neurosci. 2022 Apr;25(4):458-473
1J:246098 Dyson JM, Conduit SE, Feeney SJ, Hakim S, DiTommaso T, Fulcher AJ, Sriratana A, Ramm G, Horan KA, Gurung R, Wicking C, Smyth I, Mitchell CA, INPP5E regulates phosphoinositide-dependent cilia transition zone function. J Cell Biol. 2017 Jan 02;216(1):247-263
1J:232111 Emechebe U, Kumar P P, Rozenberg JM, Moore B, Firment A, Mirshahi T, Moon AM, T-box3 is a ciliary protein and regulates stability of the Gli3 transcription factor to control digit number. Elife. 2016;5:e07897
1J:350576 Engelhardt D, Marean A, McKean D, Petersen J, Niswander L, RSG1 is required for cilia-dependent neural tube closure. Genesis. 2024 Jun;62(3):e23602
3J:236794 Ezratty EJ, Pasolli HA, Fuchs E, A Presenilin-2-ARF4 trafficking axis modulates Notch signaling during epidermal differentiation. J Cell Biol. 2016 Jul 4;214(1):89-101
1J:283254 Ferent J, Constable S, Gigante ED, Yam PT, Mariani LE, Legue E, Liem KF Jr, Caspary T, Charron F, The Ciliary Protein Arl13b Functions Outside of the Primary Cilium in Shh-Mediated Axon Guidance. Cell Rep. 2019 Dec 10;29(11):3356-3366.e3
1J:305200 Fernandez V, Martinez-Martinez MA, Prieto-Colomina A, Cardenas A, Soler R, Dori M, Tomasello U, Nomura Y, Lopez-Atalaya JP, Calegari F, Borrell V, Repression of Irs2 by let-7 miRNAs is essential for homeostasis of the telencephalic neuroepithelium. EMBO J. 2020 Nov 2;39(21):e105479
1J:290813 Fiore L, Takata N, Acosta S, Ma W, Pandit T, Oxendine M, Oliver G, Optic vesicle morphogenesis requires primary cilia. Dev Biol. 2020 Jun 15;462(2):119-128
1J:345876 Fitzsimons LA, Tasouri E, Willaredt MA, Stetson D, Gojak C, Kirsch J, Gardner HAR, Gorgas K, Tucker KL, Primary cilia are critical for tracheoesophageal septation. Dev Dyn. 2024 Mar;253(3):312-332
1J:242328 Foerster P, Daclin M, Asm S, Faucourt M, Boletta A, Genovesio A, Spassky N, mTORC1 signaling and primary cilia are required for brain ventricle morphogenesis. Development. 2017 Jan 15;144(2):201-210
1J:214665 Fuller K, O'Connell JT, Gordon J, Mauti O, Eggenschwiler J, Rab23 regulates Nodal signaling in vertebrate left-right patterning independently of the Hedgehog pathway. Dev Biol. 2014 Jul 15;391(2):182-95
5J:176174 Garcia-Gonzalo FR, Corbit KC, Sirerol-Piquer MS, Ramaswami G, Otto EA, Noriega TR, Seol AD, Robinson JF, Bennett CL, Josifova DJ, Garcia-Verdugo JM, Katsanis N, Hildebrandt F, Reiter JF, A transition zone complex regulates mammalian ciliogenesis and ciliary membrane composition. Nat Genet. 2011 Aug;43(8):776-84
3J:228881 Gazea M, Tasouri E, Tolve M, Bosch V, Kabanova A, Gojak C, Kurtulmus B, Novikov O, Spatz J, Pereira G, Hubner W, Brodski C, Tucker KL, Blaess S, Primary cilia are critical for Sonic hedgehog-mediated dopaminergic neurogenesis in the embryonic midbrain. Dev Biol. 2016 Jan 1;409(1):55-71
2J:199499 Gerhardt C, Lier JM, Kuschel S, Ruther U, The ciliary protein Ftm is required for ventricular wall and septal development. PLoS One. 2013;8(2):e57545
1*J:262122 Gigante ED, Long AB, Ben-Ami J, Caspary T, Hypomorphic Smo mutant with inefficient ciliary enrichment disrupts the highest level of vertebrate Hedgehog response. Dev Biol. 2018 May 15;437(2):152-162
2*J:287091 Gigante ED, Taylor MR, Ivanova AA, Kahn RA, Caspary T, ARL13B regulates Sonic hedgehog signaling from outside primary cilia. Elife. 2020 Mar 4;9:e50434
5J:315450 Giridhar PV, Bell SM, Sridharan A, Rajavelu P, Kitzmiller JA, Na CL, Kofron M, Brandt EB, Ericksen M, Naren AP, Moon C, Khurana Hershey GK, Whitsett JA, Airway Epithelial KIF3A Regulates Th2 Responses to Aeroallergens. J Immunol. 2016 Dec 1;197(11):4228-4239
1J:246522 Goetz SC, Bangs F, Barrington CL, Katsanis N, Anderson KV, The Meckel syndrome- associated protein MKS1 functionally interacts with components of the BBSome and IFT complexes to mediate ciliary trafficking and hedgehog signaling. PLoS One. 2017;12(3):e0173399
1J:197579 Goetz SC, Liem KF Jr, Anderson KV, The spinocerebellar ataxia-associated gene Tau tubulin kinase 2 controls the initiation of ciliogenesis. Cell. 2012 Nov 9;151(4):847-58
1J:220469 Grego-Bessa J, Hildebrand J, Anderson KV, Morphogenesis of the mouse neural plate depends on distinct roles of cofilin 1 in apical and basal epithelial domains. Development. 2015 Apr 1;142(7):1305-14
2J:311537 Guo L, Beck T, Fulmer D, Ramos-Ortiz S, Glover J, Wang C, Moore K, Gensemer C, Morningstar J, Moore R, Schott JJ, Le Tourneau T, Koren N, Norris RA, DZIP1 regulates mammalian cardiac valve development through a Cby1-beta-catenin mechanism. Dev Dyn. 2021 Oct;250(10):1432-1449
1J:271615 Haag N, Schuler S, Nietzsche S, Hubner CA, Strenzke N, Qualmann B, Kessels MM, The Actin Nucleator Cobl Is Critical for Centriolar Positioning, Postnatal Planar Cell Polarity Refinement, and Function of the Cochlea. Cell Rep. 2018 Aug 28;24(9):2418-2431.e6
1J:344557 Hall ET, Dillard ME, Cleverdon ER, Zhang Y, Daly CA, Ansari SS, Wakefield R, Stewart DP, Pruett-Miller SM, Lavado A, Carisey AF, Johnson A, Wang YD, Selner E, Tanes M, Ryu YS, Robinson CG, Steinberg J, Ogden SK, Cytoneme signaling provides essential contributions to mammalian tissue patterning. Cell. 2024 Jan 18;187(2):276-293.e23
2*J:324299 Hamada N, Noda M, Ito H, Iwamoto I, Nagata KI, Expression Analyses of Cep152, a Responsible Gene Product for Autosomal Recessive Primary Microcephaly, during Mouse Brain Development. Dev Neurosci. 2022;44(3):162-170
1J:298587 Hasenpusch-Theil K, Laclef C, Colligan M, Fitzgerald E, Howe K, Carroll E, Abrams SR, Reiter JF, Schneider-Maunoury S, Theil T, A transient role of the ciliary gene Inpp5e in controlling direct versus indirect neurogenesis in cortical development. Elife. 2020 Aug 25;9:e58162
1J:354192 He H, Bell SM, Davis AK, Zhao S, Sridharan A, Na CL, Guo M, Xu Y, Snowball J, Swarr DT, Zacharias WJ, Whitsett JA, PRDM3/16 regulate chromatin accessibility required for NKX2-1 mediated alveolar epithelial differentiation and function. Nat Commun. 2024 Sep 16;15(1):8112
1J:255415 He M, Ye W, Wang WJ, Sison ES, Jan YN, Jan LY, Cytoplasmic Cl(-) couples membrane remodeling to epithelial morphogenesis. Proc Natl Acad Sci U S A. 2017 Dec 26;114(52):E11161-E11169
1*J:154370 Heydeck W, Zeng H, Liu A, Planar cell polarity effector gene Fuzzy regulates cilia formation and Hedgehog signal transduction in mouse. Dev Dyn. 2009 Oct 29;238(12):3035-3042
7J:203743 Higginbotham H, Guo J, Yokota Y, Umberger NL, Su CY, Li J, Verma N, Hirt J, Ghukasyan V, Caspary T, Anton ES, Arl13b-regulated cilia activities are essential for polarized radial glial scaffold formation. Nat Neurosci. 2013 Aug;16(8):1000-7
1J:331486 Hirose T, Sugitani Y, Kurihara H, Kazama H, Kusaka C, Noda T, Takahashi H, Ohno S, PAR3 restricts the expansion of neural precursor cells by regulating hedgehog signaling. Development. 2022 Nov 1;149(21):dev199931
2J:342124 Hoi KK, Xia W, Wei MM, Ulloa Navas MJ, Garcia Verdugo JM, Nachury MV, Reiter JF, Fancy SPJ, Primary cilia control oligodendrocyte precursor cell proliferation in white matter injury via Hedgehog-independent CREB signaling. Cell Rep. 2023 Oct 31;42(10):113272
1J:236236 Hong CJ, Hamilton BA, Zfp423 Regulates Sonic Hedgehog Signaling via Primary Cilium Function. PLoS Genet. 2016 Oct;12(10):e1006357
1J:309463 Hor CHH, Lo JCW, Cham ALS, Leong WY, Goh ELK, Multifaceted Functions of Rab23 on Primary Cilium-Mediated and Hedgehog Signaling-Mediated Cerebellar Granule Cell Proliferation. J Neurosci. 2021 Aug 11;41(32):6850-6863
3J:173637 Horner VL, Caspary T, Disrupted dorsal neural tube BMP signaling in the cilia mutant Arl13b hnn stems from abnormal Shh signaling. Dev Biol. 2011 Jul 1;355(1):43-54
1J:302131 Hu HB, Song ZQ, Song GP, Li S, Tu HQ, Wu M, Zhang YC, Yuan JF, Li TT, Li PY, Xu YL, Shen XL, Han QY, Li AL, Zhou T, Chun J, Zhang XM, Li HY, LPA signaling acts as a cell-extrinsic mechanism to initiate cilia disassembly and promote neurogenesis. Nat Commun. 2021 Jan 28;12(1):662
1J:160335 Huang X, Liu J, Ketova T, Fleming JT, Grover VK, Cooper MK, Litingtung Y, Chiang C, Transventricular delivery of Sonic hedgehog is essential to cerebellar ventricular zone development. Proc Natl Acad Sci U S A. 2010 May 4;107(18):8422-7
1J:310417 Hwang SH, Somatilaka BN, White K, Mukhopadhyay S, Ciliary and extraciliary Gpr161 pools repress hedgehog signaling in a tissue-specific manner. Elife. 2021 Aug 4;10:e67121
3J:253972 Hwang SH, White KA, Somatilaka BN, Shelton JM, Richardson JA, Mukhopadhyay S, The G protein-coupled receptor Gpr161 regulates forelimb formation, limb patterning and skeletal morphogenesis in a primary cilium-dependent manner. Development. 2018 Jan 8;145(1):dev154054
4J:219422 Insolera R, Bazzi H, Shao W, Anderson KV, Shi SH, Cortical neurogenesis in the absence of centrioles. Nat Neurosci. 2014 Nov;17(11):1528-35
1*J:215665 Insolera R, Shao W, Airik R, Hildebrandt F, Shi SH, SDCCAG8 regulates pericentriolar material recruitment and neuronal migration in the developing cortex. Neuron. 2014 Aug 20;83(4):805-22
1*J:334018 Janeckova E, Feng J, Guo T, Han X, Ghobadi A, Araujo-Villalba A, Rahman MS, Ziaei H, Ho TV, Pareek S, Alvarez J, Chai Y, Canonical Wnt signaling regulates soft palate development by mediating ciliary homeostasis. Development. 2023 Mar 1;150(5):dev201189
2J:250399 Jayaraman D, Kodani A, Gonzalez DM, Mancias JD, Mochida GH, Vagnoni C, Johnson J, Krogan N, Harper JW, Reiter JF, Yu TW, Bae BI, Walsh CA, Microcephaly Proteins Wdr62 and Aspm Define a Mother Centriole Complex Regulating Centriole Biogenesis, Apical Complex, and Cell Fate. Neuron. 2016 Nov 23;92(4):813-828
1J:336512 Knapp L, Sun H, Wang YM, Chen BJ, Lin X, Gao N, Chen P, Ren D, Rab11a Is Essential for the Development and Integrity of the Stereocilia and Kinocilia in the Mammalian Organ of Corti. eNeuro. 2023 Jun;10(6):ENEURO.0420-22.2023
1*J:158583 Ko HW, Norman RX, Tran J, Fuller KP, Fukuda M, Eggenschwiler JT, Broad-minded links cell cycle-related kinase to cilia assembly and hedgehog signal transduction. Dev Cell. 2010 Feb 16;18(2):237-47
1J:263095 Koefoed K, Skat-Rordam J, Andersen P, Warzecha CB, Pye M, Andersen TA, Ajbro KD, Bendsen E, Narimatsu M, Vilhardt F, Pedersen LB, Wrana JL, Anderson RH, Mollgard K, Christensen ST, Larsen LA, The E3 ubiquitin ligase SMURF1 regulates cell-fate specification and outflow tract septation during mammalian heart development. Sci Rep. 2018 Jun 22;8(1):9542
2J:221657 Kong JH, Yang L, Dessaud E, Chuang K, Moore DM, Rohatgi R, Briscoe J, Novitch BG, Notch activity modulates the responsiveness of neural progenitors to sonic hedgehog signaling. Dev Cell. 2015 May 26;33(4):373-87
3*J:311933 Kong JH, Young CB, Pusapati GV, Espinoza FH, Patel CB, Beckert F, Ho S, Patel BB, Gabriel GC, Aravind L, Bazan JF, Gunn TM, Lo CW, Rohatgi R, Gene-teratogen interactions influence the penetrance of birth defects by altering Hedgehog signaling strength. Development. 2021 Oct 1;148(19):dev199867
1J:299749 Kong JH, Young CB, Pusapati GV, Patel CB, Ho S, Krishnan A, Lin JI, Devine W, Moreau de Bellaing A, Athni TS, Aravind L, Gunn TM, Lo CW, Rohatgi R, A Membrane-Tethered Ubiquitination Pathway Regulates Hedgehog Signaling and Heart Development. Dev Cell. 2020 Nov 23;55(4):432-449.e12
1J:343556 Kretschmer V, Schneider S, Matthiessen PA, Reichert D, Hotaling N, Glassser G, Lieberwirth I, Bharti K, De Cegli R, Conte I, Nandrot EF, May-Simera HL, Deletion of IFT20 exclusively in the RPE ablates primary cilia and leads to retinal degeneration. PLoS Biol. 2023 Dec;21(12):e3002402
1J:307969 Kumar D, Rains A, Herranz-Perez V, Lu Q, Shi X, Swaney DL, Stevenson E, Krogan NJ, Huang B, Westlake C, Garcia-Verdugo JM, Yoder BK, Reiter JF, A ciliopathy complex builds distal appendages to initiate ciliogenesis. J Cell Biol. 2021 Sep 6;220(9)
4J:225435 Laclef C, Anselme I, Besse L, Catala M, Palmyre A, Baas D, Paschaki M, Pedraza M, Metin C, Durand B, Schneider-Maunoury S, The role of primary cilia in corpus callosum formation is mediated by production of the Gli3 repressor. Hum Mol Genet. 2015 Sep 1;24(17):4997-5014
1J:172420 Lancaster MA, Gopal DJ, Kim J, Saleem SN, Silhavy JL, Louie CM, Thacker BE, Williams Y, Zaki MS, Gleeson JG, Defective Wnt-dependent cerebellar midline fusion in a mouse model of Joubert syndrome. Nat Med. 2011 Jun;17(6):726-31
2J:174986 Lancaster MA, Schroth J, Gleeson JG, Subcellular spatial regulation of canonical Wnt signalling at the primary cilium. Nat Cell Biol. 2011 Jun;13(6):700-7
1J:317151 Lavado A, Gangwar R, Pare J, Wan S, Fan Y, Cao X, YAP/TAZ maintain the proliferative capacity and structural organization of radial glial cells during brain development. Dev Biol. 2021 Dec;480:39-49
1J:313459 Lee B, Panda S, Lee HY, Primary Ciliary Deficits in the Dentate Gyrus of Fragile X Syndrome. Stem Cell Reports. 2020 Aug 11;15(2):454-466
2*J:306217 Lee H, Ko HW, Cell cycle-related kinase is a crucial regulator for ciliogenesis and Hedgehog signaling in embryonic mouse lung development. BMB Rep. 2020 Jul;53(7):367-372
2*J:331469 Lee H, Moon KH, Song J, Je S, Bok J, Ko HW, Tissue-specific requirement of sodium channel and clathrin linker 1 (Sclt1) for ciliogenesis during limb development. Front Cell Dev Biol. 2022;10:1058895
1J:338829 Lee J, Kim Y, Ataliotis P, Kim HG, Kim DW, Bennett DC, Brown NA, Layman LC, Kim SH, Coordination of canonical and noncanonical Hedgehog signalling pathways mediated by WDR11 during primordial germ cell development. Sci Rep. 2023 Jul 29;13(1):12309
1J:165744 Lee JD, Migeotte I, Anderson KV, Left-right patterning in the mouse requires Epb4.1l5-dependent morphogenesis of the node and midline. Dev Biol. 2010 Oct 15;346(2):237-46
1J:345473 Lee JG, Yon JM, Kim G, Lee SG, Kim CY, Cheong SA, Kim HY, Yu J, Kim K, Sung YH, Yoo HJ, Woo DC, Rho JK, Ha CH, Pack CG, Oh SH, Lim JS, Han YM, Hong EJ, Seong JK, Lee HW, Lee SW, Lee KU, Kim CJ, Nam SY, Cho YS, Baek IJ, PIBF1 regulates trophoblast syncytialization and promotes cardiovascular development. Nat Commun. 2024 Feb 19;15(1):1487
3J:284006 Legue E, Liem KF Jr, Tulp3 Is a Ciliary Trafficking Gene that Regulates Polycystic Kidney Disease. Curr Biol. 2019 Mar 4;29(5):803-812.e5
1J:280465 Lewis WR, Bales KL, Revell DZ, Croyle MJ, Engle SE, Song CJ, Malarkey EB, Uytingco CR, Shan D, Antonellis PJ, Nagy TR, Kesterson RA, Mrug MM, Martens JR, Berbari NF, Gross AK, Yoder BK, Mks6 mutations reveal tissue- and cell type-specific roles for the cilia transition zone. FASEB J. 2019 Jan;33(1):1440-1455
3J:321504 Lex RK, Zhou W, Ji Z, Falkenstein KN, Schuler KE, Windsor KE, Kim JD, Ji H, Vokes SA, GLI transcriptional repression is inert prior to Hedgehog pathway activation. Nat Commun. 2022 Feb 10;13(1):808
1J:341535 Li L, Li J, Yuan L, A direct interaction between CENTLEIN and RABIN8 is required for primary cilium formation. Acta Biochim Biophys Sin (Shanghai). 2023 Jul 20;55(9):1434-1444
2J:327070 Li Q, Han Z, Singh N, Terre B, Fame RM, Arif U, Page TD, Zahran T, Abdeltawab A, Huang Y, Cao P, Wang J, Lu H, Lidov HGW, Surendran K, Wu L, Virga JQ, Zhao YT, Schuller U, Wechsler-Reya RJ, Lehtinen MK, Roy S, Liu Z, Stracker TH, Zhao H, Disruption of GMNC-MCIDAS multiciliogenesis program is critical in choroid plexus carcinoma development. Cell Death Differ. 2022 Aug;29(8):1596-1610
1J:185270 Liem KF Jr, Ashe A, He M, Satir P, Moran J, Beier D, Wicking C, Anderson KV, The IFT-A complex regulates Shh signaling through cilia structure and membrane protein trafficking. J Cell Biol. 2012 Jun 11;197(6):789-800
2J:292113 Lin YN, Lee YS, Li SK, Tang TK, Loss of CPAP in developing mouse brain and its functional implication for human primary microcephaly. J Cell Sci. 2020 Jun 24;133(12):jcs243592
2J:231070 Liu Y, Snedecor ER, Choi YJ, Yang N, Zhang X, Xu Y, Han Y, Jones EC, Shroyer KR, Clark RA, Zhang L, Qin C, Chen J, Gorab Is Required for Dermal Condensate Cells to Respond to Hedgehog Signals during Hair Follicle Morphogenesis. J Invest Dermatol. 2016 Feb;136(2):378-86
1J:317268 Liu Y, Yuan Q, Wang Z, Ding L, Kong N, Liu J, Hu Y, Zhang Y, Li C, Yan G, Jiang Y, Sun H, A high level of KLF12 causes folic acid-resistant neural tube defects by activating the Shh signaling pathway in mice. Biol Reprod. 2021 Oct 11;105(4):837-845
2J:343069 Lodge EJ, Barrell WB, Liu KJ, Andoniadou CL, The Fuzzy planar cell polarity protein (FUZ), necessary for primary cilium formation, is essential for pituitary development. J Anat. 2023 Oct 4;
1J:346057 Lu D, Zhi Y, Su H, Lin X, Lin J, Shi Y, Yi W, Hong C, Zhang T, Fu Z, Chen LY, Zhao Z, Li R, Xu Z, Chen W, Wang N, Xu D, ESCRT-I protein UBAP1 controls ventricular expansion and cortical neurogenesis via modulating adherens junctions of radial glial cells. Cell Rep. 2024 Feb 24;43(3):113818
1J:257916 Lupu FI, Burnett JB, Eggenschwiler JT, Cell cycle-related kinase regulates mammalian eye development through positive and negative regulation of the Hedgehog pathway. Dev Biol. 2018 Feb 1;434(1):24-35
1J:310346 Mecklenburg N, Kowalczyk I, Witte F, Gorne J, Laier A, Mamo TM, Gonschior H, Lehmann M, Richter M, Sporbert A, Purfurst B, Hubner N, Hammes A, Identification of disease-relevant modulators of the SHH pathway in the developing brain. Development. 2021 Sep 1;148(17):dev199307
2J:241005 Millington G, Elliott KH, Chang YT, Chang CF, Dlugosz A, Brugmann SA, Cilia-dependent GLI processing in neural crest cells is required for tongue development. Dev Biol. 2017 Apr 15;424(2):124-137
1J:211652 Moon H, Song J, Shin JO, Lee H, Kim HK, Eggenschwiller JT, Bok J, Ko HW, Intestinal cell kinase, a protein associated with endocrine-cerebro-osteodysplasia syndrome, is a key regulator of cilia length and Hedgehog signaling. Proc Natl Acad Sci U S A. 2014 Jun 10;111(23):8541-6
3J:308270 Moon KH, Ma JH, Min H, Koo H, Kim H, Ko HW, Bok J, Dysregulation of sonic hedgehog signaling causes hearing loss in ciliopathy mouse models. Elife. 2020 Dec 31;9:e56551
1J:338207 Moreau MX, Saillour Y, Elorriaga V, Bouloudi B, Delberghe E, Deutsch Guerrero T, Ochandorena-Saa A, Maeso-Alonso L, Marques MM, Marin MC, Spassky N, Pierani A, Causeret F, Repurposing of the multiciliation gene regulatory network in fate specification of Cajal-Retzius neurons. Dev Cell. 2023 Jun 7;
1J:193395 Mukhopadhyay S, Wen X, Ratti N, Loktev A, Rangell L, Scales SJ, Jackson PK, The ciliary G-protein-coupled receptor Gpr161 negatively regulates the Sonic hedgehog pathway via cAMP signaling. Cell. 2013 Jan 17;152(1-2):210-23
1J:278973 Munoz-Estrada J, Ferland RJ, Ahi1 promotes Arl13b ciliary recruitment, regulates Arl13b stability and is required for normal cell migration. J Cell Sci. 2019 Sep 4;132(17):jcs230680
2J:326359 Mytlis A, Kumar V, Qiu T, Deis R, Hart N, Levy K, Masek M, Shawahny A, Ahmad A, Eitan H, Nather F, Adar-Levor S, Birnbaum RY, Elia N, Bachmann-Gagescu R, Roy S, Elkouby YM, Control of meiotic chromosomal bouquet and germ cell morphogenesis by the zygotene cilium. Science. 2022 Jun 17;376(6599):eabh3104
1J:297063 Najarro EH, Huang J, Jacobo A, Quiruz LA, Grillet N, Cheng AG, Dual regulation of planar polarization by secreted Wnts and Vangl2 in the developing mouse cochlea. Development. 2020 Oct 5;147(19):dev191981
3J:309437 Nakagawa N, Li J, Yabuno-Nakagawa K, Eom TY, Cowles M, Mapp T, Taylor R, Anton ES, APC sets the Wnt tone necessary for cerebral cortical progenitor development. Genes Dev. 2017 Aug 15;31(16):1679-1692
1J:273907 Nandadasa S, Kraft CM, Wang LW, O'Donnell A, Patel R, Gee HY, Grobe K, Cox TC, Hildebrandt F, Apte SS, Secreted metalloproteases ADAMTS9 and ADAMTS20 have a non-canonical role in ciliary vesicle growth during ciliogenesis. Nat Commun. 2019 Feb 27;10(1):953
1J:285992 Ono K, Keller J, Lopez Ramirez O, Gonzalez Garrido A, Zobeiri OA, Chang HHV, Vijayakumar S, Ayiotis A, Duester G, Della Santina CC, Jones SM, Cullen KE, Eatock RA, Wu DK, Retinoic acid degradation shapes zonal development of vestibular organs and sensitivity to transient linear accelerations. Nat Commun. 2020 Jan 2;11(1):63
1J:235630 Paronett EM, Meechan DW, Karpinski BA, LaMantia AS, Maynard TM, Ranbp1, Deleted in DiGeorge/22q11.2 Deletion Syndrome, is a Microcephaly Gene That Selectively Disrupts Layer 2/3 Cortical Projection Neuron Generation. Cereb Cortex. 2015 Oct;25(10):3977-93
1J:196506 Pasek RC, Berbari NF, Lewis WR, Kesterson RA, Yoder BK, Mammalian Clusterin associated protein 1 is an evolutionarily conserved protein required for ciliogenesis. Cilia. 2012;1(1):20
8J:317613 Patnaik SR, Kretschmer V, Brucker L, Schneider S, Volz AK, Oancea-Castillo LDR, May-Simera HL, Bardet-Biedl Syndrome proteins regulate cilia disassembly during tissue maturation. Cell Mol Life Sci. 2019 Feb;76(4):757-775
2J:308707 Paulson D, Harms R, Ward C, Latterell M, Pazour GJ, Fink DM, Loss of Primary Cilia Protein IFT20 Dysregulates Lymphatic Vessel Patterning in Development and Inflammation. Front Cell Dev Biol. 2021;9:672625
1J:306169 Peralta M, Ortiz Lopez L, Jerabkova K, Lucchesi T, Vitre B, Han D, Guillemot L, Dingare C, Sumara I, Mercader N, Lecaudey V, Delaval B, Meilhac SM, Vermot J, Intraflagellar Transport Complex B Proteins Regulate the Hippo Effector Yap1 during Cardiogenesis. Cell Rep. 2020 Jul 21;32(3):107932
1J:291752 Portal C, Rompolas P, Lwigale P, Iomini C, Primary cilia deficiency in neural crest cells models anterior segment dysgenesis in mouse. Elife. 2019 Dec 17;8:e52423
1J:214731 Pulina M, Liang D, Astrof S, Shape and position of the node and notochord along the bilateral plane of symmetry are regulated by cell-extracellular matrix interactions. Biol Open. 2014;3(7):583-90
1J:168317 Qin J, Lin Y, Norman RX, Ko HW, Eggenschwiler JT, Intraflagellar transport protein 122 antagonizes Sonic Hedgehog signaling and controls ciliary localization of pathway components. Proc Natl Acad Sci U S A. 2011 Jan 25;108(4):1456-61
2J:243429 Rakotomamonjy J, Brunner M, Juschke C, Zang K, Huang EJ, Reichardt LF, Chenn A, Afadin controls cell polarization and mitotic spindle orientation in developing cortical radial glia. Neural Dev. 2017 May 08;12(1):7
1J:238551 Ramkumar N, Omelchenko T, Silva-Gagliardi NF, McGlade CJ, Wijnholds J, Anderson KV, Crumbs2 promotes cell ingression during the epithelial-to-mesenchymal transition at gastrulation. Nat Cell Biol. 2016 Dec;18(12):1281-1291
1J:295179 Ramos SI, Makeyev EV, Salierno M, Kodama T, Kawakami Y, Sahara S, Tuba8 Drives Differentiation of Cortical Radial Glia into Apical Intermediate Progenitors by Tuning Modifications of Tubulin C Termini. Dev Cell. 2020 Feb 24;52(4):477-491.e8
2J:314161 Rashidieh B, Shohayeb B, Bain AL, Fortuna PRJ, Sinha D, Burgess A, Mills R, Adams RC, Lopez JA, Blumbergs P, Finnie J, Kalimutho M, Piper M, Hudson JE, Ng DCH, Khanna KK, Cep55 regulation of PI3K/Akt signaling is required for neocortical development and ciliogenesis. PLoS Genet. 2021 Oct;17(10):e1009334
1J:233509 Roberson EC, Dowdle WE, Ozanturk A, Garcia-Gonzalo FR, Li C, Halbritter J, Elkhartoufi N, Porath JD, Cope H, Ashley-Koch A, Gregory S, Thomas S, Sayer JA, Saunier S, Otto EA, Katsanis N, Davis EE, Attie-Bitach T, Hildebrandt F, Leroux MR, Reiter JF, TMEM231, mutated in orofaciodigital and Meckel syndromes, organizes the ciliary transition zone. J Cell Biol. 2015 Apr 13;209(1):129-42
1J:278197 Robson A, Makova SZ, Barish S, Zaidi S, Mehta S, Drozd J, Jin SC, Gelb BD, Seidman CE, Chung WK, Lifton RP, Khokha MK, Brueckner M, Histone H2B monoubiquitination regulates heart development via epigenetic control of cilia motility. Proc Natl Acad Sci U S A. 2019 Jul 9;116(28):14049-14054
1*J:173396 Sang L, Miller JJ, Corbit KC, Giles RH, Brauer MJ, Otto EA, Baye LM, Wen X, Scales SJ, Kwong M, Huntzicker EG, Sfakianos MK, Sandoval W, Bazan JF, Kulkarni P, Garcia-Gonzalo FR, Seol AD, O'Toole JF, Held S, Reutter HM, Lane WS, Rafiq MA, Noor A, Ansar M,Devi AR, Sheffield VC, Slusarski DC, Vincent JB, Doherty DA, Hildebrandt F, Reiter JF, Jackson PK, Mapping the NPHP-JBTS-MKS protein network reveals ciliopathy disease genes and pathways. Cell. 2011 May 13;145(4):513-28
1J:250948 Schock EN, Brugmann SA, Neural crest cells utilize primary cilia to regulate ventral forebrain morphogenesis via Hedgehog-dependent regulation of oriented cell division. Dev Biol. 2017 Nov 15;431(2):168-178
2J:241357 Schock EN, Struve JN, Chang CF, Williams TJ, Snedeker J, Attia AC, Stottmann RW, Brugmann SA, A tissue-specific role for intraflagellar transport genes during craniofacial development. PLoS One. 2017;12(3):e0174206
1J:346597 Seo S, Sonn SK, Kweon HY, Jin J, Kume T, Ko JY, Park JH, Oh GT, Primary Cilium in Neural Crest Cells Crucial for Anterior Segment Development and Corneal Avascularity. Invest Ophthalmol Vis Sci. 2024 Mar 5;65(3):30
3J:293277 Shao W, Yang J, He M, Yu XY, Lee CH, Yang Z, Joyner AL, Anderson KV, Zhang J, Tsou MB, Shi H, Shi SH, Centrosome anchoring regulates progenitor properties and cortical formation. Nature. 2020 Apr;580(7801):106-112
2J:346646 Sharma R, Kalot R, Levin Y, Babayeva S, Kachurina N, Chung CF, Liu KJ, Bouchard M, Torban E, The CPLANE protein Fuzzy regulates ciliogenesis by suppressing actin polymerization at the base of the primary cilium via p190A RhoGAP. Development. 2024 Mar 15;151(6):dev202322
2J:270726 Shimada IS, Hwang SH, Somatilaka BN, Wang X, Skowron P, Kim J, Kim M, Shelton JM, Rajaram V, Xuan Z, Taylor MD, Mukhopadhyay S, Basal Suppression of the Sonic Hedgehog Pathway by the G-Protein-Coupled Receptor Gpr161 Restricts Medulloblastoma Pathogenesis. Cell Rep. 2018 Jan 30;22(5):1169-1184
2J:276848 Shimada IS, Somatilaka BN, Hwang SH, Anderson AG, Shelton JM, Rajaram V, Konopka G, Mukhopadhyay S, Derepression of sonic hedgehog signaling upon Gpr161 deletion unravels forebrain and ventricular abnormalities. Dev Biol. 2019 Jun 1;450(1):47-62
2J:285212 Shohayeb B, Ho U, Yeap YY, Parton RG, Millard SS, Xu Z, Piper M, Ng DCH, The association of microcephaly protein WDR62 with CPAP/IFT88 is required for cilia formation and neocortical development. Hum Mol Genet. 2020 Jan 15;29(2):248-263
1J:312425 Shukla S, Haenold R, Urbanek P, Frappart L, Monajembashi S, Grigaravicius P, Nagel S, Min WK, Tapias A, Kassel O, Heuer H, Wang ZQ, Ploubidou A, Herrlich P, TRIP6 functions in brain ciliogenesis. Nat Commun. 2021 Oct 7;12(1):5887
2*J:286656 Shylo NA, Emmanouil E, Ramrattan D, Weatherbee SD, Loss of ciliary transition zone protein TMEM107 leads to heterotaxy in mice. Dev Biol. 2020 Apr 15;460(2):187-199
1J:255142 Siller SS, Sharma H, Li S, Yang J, Zhang Y, Holtzman MJ, Winuthayanon W, Colognato H, Holdener BC, Li FQ, Takemaru KI, Conditional knockout mice for the distal appendage protein CEP164 reveal its essential roles in airway multiciliated cell differentiation. PLoS Genet. 2017 Dec;13(12):e1007128
2J:275391 Simmons AJ, Park R, Sterling NA, Jang MH, van Deursen JMA, Yen TJ, Cho SH, Kim S, Nearly complete deletion of BubR1 causes microcephaly through shortened mitosis and massive cell death. Hum Mol Genet. 2019 Jun 1;28(11):1822-1836
2J:243907 Snouffer A, Brown D, Lee H, Walsh J, Lupu F, Norman R, Lechtreck K, Ko HW, Eggenschwiler J, Cell Cycle-Related Kinase (CCRK) regulates ciliogenesis and Hedgehog signaling in mice. PLoS Genet. 2017 Aug;13(8):e1006912
3J:297055 Somatilaka BN, Hwang SH, Palicharla VR, White KA, Badgandi H, Shelton JM, Mukhopadhyay S, Ankmy2 Prevents Smoothened-Independent Hyperactivation of the Hedgehog Pathway via Cilia-Regulated Adenylyl Cyclase Signaling. Dev Cell. 2020 Sep 28;54(6):710-726.e8
1J:239367 Stasiulewicz M, Gray SD, Mastromina I, Silva JC, Bjorklund M, Seymour PA, Booth D, Thompson C, Green RJ, Hall EA, Serup P, Dale JK, A conserved role for Notch signaling in priming the cellular response to Shh through ciliary localisation of the key Shh transducer Smo. Development. 2015 Jul 01;142(13):2291-303
1J:298840 Sterling N, Duncan AR, Park R, Koolen DA, Shi J, Cho SH, Benke PJ, Grant PE, Genetti CA, VanNoy GE, Juusola J, McWalter K, Parboosingh JS, Lamont RE, Bernier FP, Smith C, Harris DJ, Stegmann APA, Innes AM, Kim S, Agrawal PB, De novo variants in MPP5 cause global developmental delay and behavioral changes. Hum Mol Genet. 2020 Dec 18;29(20):3388-3401
1J:232342 Stewart K, Gaitan Y, Shafer ME, Aoudjit L, Hu D, Sharma R, Tremblay M, Ishii H, Marcotte M, Stanga D, Tang YC, Boualia SK, Nguyen AH, Takano T, Lamarche-Vane N, Vidal S, Bouchard M, A Point Mutation in p190A RhoGAP Affects Ciliogenesis and Leads to Glomerulocystic Kidney Defects. PLoS Genet. 2016 Feb;12(2):e1005785
1J:332483 Suciu SK, Long AB, Caspary T, Smoothened and ARL13B are critical in mouse for superior cerebellar peduncle targeting. Genetics. 2021;218(4)
1J:278287 Tang T, Zhang Y, Wang Y, Cai Z, Lu Z, Li L, Huang R, Hagelkruys A, Matthias P, Zhang H, Seiser C, Xie Y, HDAC1 and HDAC2 Regulate Intermediate Progenitor Positioning to Safeguard Neocortical Development. Neuron. 2019 Mar 20;101(6):1117-1133.e5
1J:294899 Terry TT, Cheng T, Mahjoub M, Zong H, Mosaic Analysis with Double Markers reveals IGF1R function in granule cell progenitors during cerebellar development. Dev Biol. 2020 Sep 15;465(2):130-143
1J:309653 Tong Y, Park SH, Wu D, Xu W, Guillot SJ, Jin L, Li X, Wang Y, Lin CS, Fu Z, An essential role of intestinal cell kinase in lung development is linked to the perinatal lethality of human ECO syndrome. FEBS Lett. 2017 May;591(9):1247-1257
3J:181006 Tuson M, He M, Anderson KV, Protein kinase A acts at the basal body of the primary cilium to prevent Gli2 activation and ventralization of the mouse neural tube. Development. 2011 Nov;138(22):4921-30
1J:288448 Uzquiano A, Cifuentes-Diaz C, Jabali A, Romero DM, Houllier A, Dingli F, Maillard C, Boland A, Deleuze JF, Loew D, Mancini GMS, Bahi-Buisson N, Ladewig J, Francis F, Mutations in the Heterotopia Gene Eml1/EML1 Severely Disrupt the Formation of Primary Cilia. Cell Rep. 2019 Aug 6;28(6):1596-1611.e10
2J:178643 Van Campenhout CA, Eitelhuber A, Gloeckner CJ, Giallonardo P, Gegg M, Oller H, Grant SG, Krappmann D, Ueffing M, Lickert H, Dlg3 trafficking and apical tight junction formation is regulated by nedd4 and nedd4-2 e3 ubiquitin ligases. Dev Cell. 2011 Sep 13;21(3):479-91
1J:332376 Van Heurck R, Bonnefont J, Wojno M, Suzuki IK, Velez-Bravo FD, Erkol E, Nguyen DT, Herpoel A, Bilheu A, Beckers S, Ledent C, Vanderhaeghen P, CROCCP2 acts as a human-specific modifier of cilia dynamics and mTOR signaling to promote expansion of cortical progenitors. Neuron. 2023 Jan 4;111(1):65-80.e6
1J:226310 Veleri S, Manjunath SH, Fariss RN, May-Simera H, Brooks M, Foskett TA, Gao C, Longo TA, Liu P, Nagashima K, Rachel RA, Li T, Dong L, Swaroop A, Ciliopathy-associated gene Cc2d2a promotes assembly of subdistal appendages on the mother centriole during cilia biogenesis. Nat Commun. 2014;5:4207
2*J:216502 Wainwright EN, Svingen T, Ng ET, Wicking C, Koopman P, Primary cilia function regulates the length of the embryonic trunk axis and urogenital field in mice. Dev Biol. 2014 Nov 15;395(2):342-54
1*J:353362 Walker MF, Zhang J, Steiner W, Ku PI, Zhu JF, Michaelson Z, Yen YC, Lee A, Long AB, Casey MJ, Poddar A, Nelson IB, Arveseth CD, Nagel F, Clough R, LaPotin S, Kwan KM, Schulz S, Stewart RA, Tesmer JJG, Caspary T, Subramanian R, Ge X, Myers BR, GRK2 kinases in the primary cilium initiate SMOOTHENED-PKA signaling in the Hedgehog cascade. PLoS Biol. 2024 Aug;22(8):e3002685
1J:190319 Walton KD, Kolterud A, Czerwinski MJ, Bell MJ, Prakash A, Kushwaha J, Grosse AS, Schnell S, Gumucio DL, Hedgehog-responsive mesenchymal clusters direct patterning and emergence of intestinal villi. Proc Natl Acad Sci U S A. 2012 Sep 25;109(39):15817-22
1J:284292 Wang B, Zhang Y, Dong H, Gong S, Wei B, Luo M, Wang H, Wu X, Liu W, Xu X, Zheng Y, Sun M, Loss of Tctn3 causes neuronal apoptosis and neural tube defects in mice. Cell Death Dis. 2018 May 1;9(5):520
1J:262219 Wang B, Zheng Y, Shi H, Du X, Zhang Y, Wei B, Luo M, Wang H, Wu X, Hua X, Sun M, Xu X, Zfp462 deficiency causes anxiety-like behaviors with excessive self-grooming in mice. Genes Brain Behav. 2017 Feb;16(2):296-307
1J:249241 Wang C, Li J, Meng Q, Wang B, Three Tctn proteins are functionally conserved in the regulation of neural tube patterning and Gli3 processing but not ciliogenesis and Hedgehog signaling in the mouse. Dev Biol. 2017 Oct 1;430(1):156-165
1*J:260445 Wang C, Li J, Takemaru KI, Jiang X, Xu G, Wang B, Centrosomal protein Dzip1l binds Cby, promotes ciliary bud formation, and acts redundantly with Bromi to regulate ciliogenesis in the mouse. Development. 2018 Mar 15;145(6):dev164236
3*J:154075 Weatherbee SD, Niswander LA, Anderson KV, A mouse model for Meckel syndrome reveals Mks1 is required for ciliogenesis and Hedgehog signaling. Hum Mol Genet. 2009 Dec 1;18(23):4565-75
2J:302983 Wildung M, Esser TU, Grausam KB, Wiedwald C, Volceanov-Hahn L, Riedel D, Beuermann S, Li L, Zylla J, Guenther AK, Wienken M, Ercetin E, Han Z, Bremmer F, Shomroni O, Andreas S, Zhao H, Lize M, Transcription factor TAp73 and microRNA-449 complement each other to support multiciliogenesis. Cell Death Differ. 2019 Dec;26(12):2740-2757
2J:196616 Willaredt MA, Gorgas K, Gardner HA, Tucker KL, Multiple essential roles for primary cilia in heart development. Cilia. 2012;1(1):23
1J:325905 Willnow D, Benary U, Margineanu A, Vignola ML, Konrath F, Pongrac IM, Karimaddini Z, Vigilante A, Wolf J, Spagnoli FM, Quantitative lineage analysis identifies a hepato-pancreato-biliary progenitor niche. Nature. 2021 Sep;597(7874):87-91
1J:242833 Wu C, Li J, Peterson A, Tao K, Wang B, Loss of dynein-2 intermediate chain Wdr34 results in defects in retrograde ciliary protein trafficking and Hedgehog signaling in the mouse. Hum Mol Genet. 2017 Jul 01;26(13):2386-2397
1J:317033 Xie C, Abrams SR, Herranz-Perez V, Garcia-Verdugo JM, Reiter JF, Endoderm development requires centrioles to restrain p53-mediated apoptosis in the absence of ERK activity. Dev Cell. 2021 Dec 20;56(24):3334-3348.e6
1J:240543 Xin D, Christopher KJ, Zeng L, Kong Y, Weatherbee SD, IFT56 regulates vertebrate developmental patterning by maintaining IFTB complex integrity and ciliary microtubule architecture. Development. 2017 Apr 15;144(8):1544-1553
1*J:267206 Xu S, Liu Y, Meng Q, Wang B, Rab34 small GTPase is required for Hedgehog signaling and an early step of ciliary vesicle formation in mouse. J Cell Sci. 2018 Nov 8;131(21):jcs213710
2*J:224085 Yang N, Li L, Eguether T, Sundberg JP, Pazour GJ, Chen J, Intraflagellar transport 27 is essential for hedgehog signaling but dispensable for ciliogenesis during hair follicle morphogenesis. Development. 2015 Jun 15;142(12):2194-202
1J:317198 Yang Y, Paivinen P, Xie C, Krup AL, Makela TP, Mostov KE, Reiter JF, Ciliary Hedgehog signaling patterns the digestive system to generate mechanical forces driving elongation. Nat Commun. 2021 Dec 10;12(1):7186
1J:352471 Yoshida S, Tsuneoka Y, Tsukada T, Nakakura T, Kawamura A, Kai W, Yoshida K, Primary Cilia are Required for Cell-Type Determination and Angiogenesis in Pituitary Development. Endocrinology. 2024 Jul 1;165(8)
1J:352479 Zeng H, Ali S, Sebastian A, Ramos-Medero AS, Albert I, Dean C, Liu A, CPLANE protein INTU regulates growth and patterning of the mouse lungs through cilia-dependent Hh signaling. Dev Biol. 2024 Jul 17;515:92-101
1J:159120 Zeng H, Hoover AN, Liu A, PCP effector gene Inturned is an important regulator of cilia formation and embryonic development in mammals. Dev Biol. 2010 Mar 15;339(2):418-28
1*J:155058 Zhang H, Ables ET, Pope CF, Washington MK, Hipkens S, Means AL, Path G, Seufert J, Costa RH, Leiter AB, Magnuson MA, Gannon M, Multiple, temporal-specific roles for HNF6 in pancreatic endocrine and ductal differentiation. Mech Dev. 2009 Dec;126(11-12):958-73
3J:333039 Zhang K, Da Silva F, Seidl C, Wilsch-Brauninger M, Herbst J, Huttner WB, Niehrs C, Primary cilia are WNT-transducing organelles whose biogenesis is controlled by a WNT-PP1 axis. Dev Cell. 2023 Jan 23;58(2):139-154.e8
1J:177881 Zhang Z, Wlodarczyk BJ, Niederreither K, Venugopalan S, Florez S, Finnell RH, Amendt BA, Fuz regulates craniofacial development through tissue specific responses to signaling factors. PLoS One. 2011;6(9):e24608
1*J:240405 Zhao L, Arsenault M, Ng ET, Longmuss E, Chau TC, Hartwig S, Koopman P, SOX4 regulates gonad morphogenesis and promotes male germ cell differentiation in mice. Dev Biol. 2017 Mar 01;423(1):46-56
1J:353625 Zhu Y, Huang Y, Tang T, Xie Y, HDAC1 and HDAC2 orchestrate Wnt signaling to regulate neural progenitor transition during brain development. iScience. 2024 Sep 20;27(9):110600

Contributing Projects:
Mouse Genome Database (MGD), Gene Expression Database (GXD), Mouse Models of Human Cancer database (MMHCdb) (formerly Mouse Tumor Biology (MTB)), Gene Ontology (GO)
Citing These Resources
Funding Information
Warranty Disclaimer, Privacy Notice, Licensing, & Copyright
Send questions and comments to User Support.
last database update
11/12/2024
MGI 6.24
The Jackson Laboratory