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David Keays

The Keays lab aims to understand two biological phenomena: 1) How do neurons migrate in the developing brain? and 2) How do animals detect magnetic fields? In tackling these two questions we adopt an interdisciplinary approach, employing a wide range of genetic, molecular, cellular, and behavioural tools.

Publications

2018

Gstrein, T., Edwards, A., Přistoupilová, A., Leca, I., Breuss, M., Pilat-Carotta, S., Hansen, AH., Tripathy, R., Traunbauer, AK., Hochstoeger, T., Rosoklija, G., Repic, M., Landler, L., Stránecký, V., Dürnberger, G., Keane, TM., Zuber, J., Adams, DJ., Flint, J., Honzik, T., Gut, M., Beltran, S., Mechtler, K., Sherr, E., Kmoch, S., Gut, I., Keays, DA. (2018) Mutations in Vps15 perturb neuronal migration in mice and are associated with neurodevelopmental disease in humans. Nat Neurosci. 21(2):207-217
Holt, C., Campbell, M., Keays, DA., Edelman, N., Kapusta, A., Maclary, E., T Domyan, E., Suh, A., Warren, WC., Yandell, M., Gilbert, MTP., Shapiro, MD. (2018) Improved Genome Assembly and Annotation for the Rock Pigeon (Columba livia). G3 (Bethesda). 8(5):1391-1398
Landler, L., Nimpf, S., Hochstoeger, T., Nordmann, GC., Papadaki-Anastasopoulou, A., Keays, DA. (2018) Comment on "Magnetosensitive neurons mediate geomagnetic orientation in Caenorhabditis elegans". Elife. 7

2017

Breuss, MW., Hansen, AH., Landler, L., Keays, DA. (2017) Brain-specific knockin of the pathogenic Tubb5 E401K allele causes defects in motor coordination and prepulse inhibition. Behav Brain Res. 323:47-55
Breuss, MW., Leca, I., Gstrein, T., Hansen, AH., Keays, DA. (2017) Tubulins and brain development - The origins of functional specification. Mol Cell Neurosci.
Breuss, MW., Nguyen, T., Srivatsan, A., Leca, I., Tian, G., Fritz, T., Hansen, AH., Musaev, D., McEvoy-Venneri, J., James, KN., Rosti, RO., Scott, E., Tan, U., Kolodner, RD., Cowan, NJ., Keays, DA., Gleeson, JG. (2017) Uner Tan syndrome caused by a homozygous TUBB2B mutation affecting microtubule stability. Hum Mol Genet. 26(2):258-269
Conti, V., Carabalona, A., Pallesi-Pocachard, E., Leventer, RJ., Schaller, F., Parrini, E., Deparis, AA., Watrin, F., Buhler, E., Novara, F., Lise, S., Pagnamenta, AT., Kini, U., Taylor, JC., Zuffardi, O., Represa, A., Keays, DA., Guerrini, R., Falace, A., Cardoso, C. (2017) A Novel Strategy Combining Array-CGH, Whole-exome Sequencing and In Utero Electroporation in Rodents to Identify Causative Genes for Brain Malformations. J Vis Exp. (130)
Nimpf, S., Keays, DA. (2017) Is magnetogenetics the new optogenetics? EMBO J. 36(12):1643-1646
Nimpf, S., Malkemper, EP., Lauwers, M., Ushakova, L., Nordmann, G., Wenninger-Weinzierl, A., Burkard, TR., Jacob, S., Heuser, T., Resch, GP., Keays, DA. (2017) Subcellular analysis of pigeon hair cells implicates vesicular trafficking in cuticulosome formation and maintenance. Elife. 6
Nordmann, GC., Hochstoeger, T., Keays, DA. (2017) Magnetoreception-A sense without a receptor. PLoS Biol. 15(10):e2003234

2016

Breuss, M., Fritz, T., Gstrein, T., Chan, K., Ushakova, L., Yu, N., Vonberg, FW., Werner, B., Elling, U., Keays, DA. (2016) Mutations in the murine homologue of TUBB5 cause microcephaly by perturbing cell cycle progression and inducing p53-associated apoptosis. Development. 143(7):1126-33
Broix, L., Jagline, H., L Ivanova, E., Schmucker, S., Drouot, N., Clayton-Smith, J., Pagnamenta, AT., Metcalfe, KA., Isidor, B., Louvier, UW., Poduri, A., Taylor, JC., Tilly, P., Poirier, K., Saillour, Y., Lebrun, N., Stemmelen, T., Rudolf, G., Muraca, G., Saintpierre, B., Elmorjani, A., Moïse, M., Weirauch, NB., Guerrini, R., Boland, A., Olaso, R., Masson, C., Tripathy, R., Keays, D., Beldjord, C., Nguyen, L., Godin, J., Kini, U., Nischké, P., Deleuze, JF., Bahi-Buisson, N., Sumara, I., Hinckelmann, MV., Chelly, J. (2016) Mutations in the HECT domain of NEDD4L lead to AKT-mTOR pathway deregulation and cause periventricular nodular heterotopia. Nat Genet. 48(11):1349-1358
Haas, MA., Ngo, L., Li, SS., Schleich, S., Qu, Z., Vanyai, HK., Cullen, HD., Cardona-Alberich, A., Gladwyn-Ng, IE., Pagnamenta, AT., Taylor, JC., Stewart, H., Kini, U., Duncan, KE., Teleman, AA., Keays, DA., Heng, JI. (2016) De Novo Mutations in DENR Disrupt Neuronal Development and Link Congenital Neurological Disorders to Faulty mRNA Translation Re-initiation. Cell Rep. 15(10):2251-65
Hempel, A., Pagnamenta, AT., Blyth, M., Mansour, S., McConnell, V., Kou, I., Ikegawa, S., Tsurusaki, Y., Matsumoto, N., Lo-Castro, A., Plessis, G., Albrecht, B., Battaglia, A., Taylor, JC., Howard, MF., Keays, D., Sohal, AS., Kühl, SJ., Kini, U., McNeill, A. (2016) Deletions and de novo mutations of SOX11 are associated with a neurodevelopmental disorder with features of Coffin-Siris syndrome. J Med Genet. 53(3):152-62
Lange, L., Pagnamenta, AT., Lise, S., Clasper, S., Stewart, H., Akha, ES., Quaghebeur, G., Knight, SJ., Keays, DA., Taylor, JC., Kini, U. (2016) A de novo frameshift in HNRNPK causing a Kabuki-like syndrome with nodular heterotopia. Clin Genet. 90(3):258-62
Pagnamenta, AT., Howard, MF., Knight, SJ., Keays, DA., Quaghebeur, G., Taylor, JC., Kini, U. (2016) Activation of an exonic splice-donor site in exon 30 of CDK5RAP2 in a patient with severe microcephaly and pigmentary abnormalities. Clin Case Rep. 4(10):952-956

2015

Breuss, M., Morandell, J., Nimpf, S., Gstrein, T., Lauwers, M., Hochstoeger, T., Braun, A., Chan, K., Sánchez Guajardo, ER., Zhang, L., Suplata, M., Heinze, KG., Elsayad, K., Keays, DA. (2015) The Expression of Tubb2b Undergoes a Developmental Transition in Murine Cortical Neurons. J Comp Neurol. 523(15):2161-86
Edelman, NB., Fritz, T., Nimpf, S., Pichler, P., Lauwers, M., Hickman, RW., Papadaki-Anastasopoulou, A., Ushakova, L., Heuser, T., Resch, GP., Saunders, M., Shaw, JA., Keays, DA. (2015) No evidence for intracellular magnetite in putative vertebrate magnetoreceptors identified by magnetic screening. Proc Natl Acad Sci U S A. 112(1):262-7
Ilkovski, B., Pagnamenta, AT., O'Grady, GL., Kinoshita, T., Howard, MF., Lek, M., Thomas, B., Turner, A., Christodoulou, J., Sillence, D., Knight, SJ., Popitsch, N., Keays, DA., Anzilotti, C., Goriely, A., Waddell, LB., Brilot, F., North, KN., Kanzawa, N., Macarthur, DG., Taylor, JC., Kini, U., Murakami, Y., Clarke, NF. (2015) Mutations in PIGY: expanding the phenotype of inherited glycosylphosphatidylinositol deficiencies. Hum Mol Genet. 24(21):6146-59
Isrie, M., Breuss, M., Tian, G., Hansen, AH., Cristofoli, F., Morandell, J., Kupchinsky, ZA., Sifrim, A., Rodriguez-Rodriguez, CM., Dapena, EP., Doonanco, K., Leonard, N., Tinsa, F., Moortgat, S., Ulucan, H., Koparir, E., Karaca, E., Katsanis, N., Marton, V., Vermeesch, JR., Davis, EE., Cowan, NJ., Keays, DA., Van Esch, H. (2015) Mutations in Either TUBB or MAPRE2 Cause Circumferential Skin Creases Kunze Type. Am J Hum Genet. 97(6):790-800
Pagnamenta, AT., Howard, MF., Wisniewski, E., Popitsch, N., Knight, SJ., Keays, DA., Quaghebeur, G., Cox, H., Cox, P., Balla, T., Taylor, JC., Kini, U. (2015) Germline recessive mutations in PI4KA are associated with perisylvian polymicrogyria, cerebellar hypoplasia and arthrogryposis. Hum Mol Genet. 24(13):3732-41

2014

Breuss, M., Keays, DA. (2014) Microtubules and neurodevelopmental disease: the movers and the makers. Adv Exp Med Biol. 800:75-96
Howard, MF., Murakami, Y., Pagnamenta, AT., Daumer-Haas, C., Fischer, B., Hecht, J., Keays, DA., Knight, SJ., Kölsch, U., Krüger, U., Leiz, S., Maeda, Y., Mitchell, D., Mundlos, S., Phillips, JA., Robinson, PN., Kini, U., Taylor, JC., Horn, D., Kinoshita, T., Krawitz, PM. (2014) Mutations in PGAP3 impair GPI-anchor maturation, causing a subtype of hyperphosphatasia with mental retardation. Am J Hum Genet. 94(2):278-87
Ngo, L., Haas, M., Qu, Z., Li, SS., Zenker, J., Teng, KS., Gunnersen, JM., Breuss, M., Habgood, M., Keays, DA., Heng, JI. (2014) TUBB5 and its disease-associated mutations influence the terminal differentiation and dendritic spine densities of cerebral cortical neurons. Hum Mol Genet. 23(19):5147-58

2013

Conti, V., Carabalona, A., Pallesi-Pocachard, E., Parrini, E., Leventer, RJ., Buhler, E., McGillivray, G., Michel, FJ., Striano, P., Mei, D., Watrin, F., Lise, S., Pagnamenta, AT., Taylor, JC., Kini, U., Clayton-Smith, J., Novara, F., Zuffardi, O., Dobyns, WB., Scheffer, IE., Robertson, SP., Berkovic, SF., Represa, A., Keays, DA., Cardoso, C., Guerrini, R. (2013) Periventricular heterotopia in 6q terminal deletion syndrome: role of the C6orf70 gene. Brain. 136(Pt 11):3378-94
Lauwers, M., Pichler, P., Edelman, NB., Resch, GP., Ushakova, L., Salzer, MC., Heyers, D., Saunders, M., Shaw, J., Keays, DA. (2013) An iron-rich organelle in the cuticular plate of avian hair cells. Curr Biol. 23(10):924-9
Treiber, CD., Salzer, M., Breuss, M., Ushakova, L., Lauwers, M., Edelman, N., Keays, DA. (2013) High resolution anatomical mapping confirms the absence of a magnetic sense system in the rostral upper beak of pigeons. Commun Integr Biol. 6(4):e24859
Watkins-Chow, DE., Cooke, J., Pidsley, R., Edwards, A., Slotkin, R., Leeds, KE., Mullen, R., Baxter, LL., Campbell, TG., Salzer, MC., Biondini, L., Gibney, G., Phan Dinh Tuy, F., Chelly, J., Morris, HD., Riegler, J., Lythgoe, MF., Arkell, RM., Loreni, F., Flint, J., Pavan, WJ., Keays, DA. (2013) Mutation of the diamond-blackfan anemia gene Rps7 in mouse results in morphological and neuroanatomical phenotypes. PLoS Genet. 9(1):e1003094

2012

Breuss, M., Heng, JI., Poirier, K., Tian, G., Jaglin, XH., Qu, Z., Braun, A., Gstrein, T., Ngo, L., Haas, M., Bahi-Buisson, N., Moutard, ML., Passemard, S., Verloes, A., Gressens, P., Xie, Y., Robson, KJ., Rani, DS., Thangaraj, K., Clausen, T., Chelly, J., Cowan, NJ., Keays, DA. (2012) Mutations in the β-tubulin gene TUBB5 cause microcephaly with structural brain abnormalities. Cell Rep. 2(6):1554-62
Pagnamenta, AT., Lise, S., Harrison, V., Stewart, H., Jayawant, S., Quaghebeur, G., Deng, AT., Murphy, VE., Sadighi Akha, E., Rimmer, A., Mathieson, I., Knight, SJ., Kini, U., Taylor, JC., Keays, DA. (2012) Exome sequencing can detect pathogenic mosaic mutations present at low allele frequencies. J Hum Genet. 57(1):70-2
Pagnamenta, AT., Murray, JE., Yoon, G., Sadighi Akha, E., Harrison, V., Bicknell, LS., Ajilogba, K., Stewart, H., Kini, U., Taylor, JC., Keays, DA., Jackson, AP., Knight, SJ. (2012) A novel nonsense CDK5RAP2 mutation in a Somali child with primary microcephaly and sensorineural hearing loss. Am J Med Genet A. 158A(10):2577-82
Treiber, CD., Salzer, MC., Riegler, J., Edelman, N., Sugar, C., Breuss, M., Pichler, P., Cadiou, H., Saunders, M., Lythgoe, M., Shaw, J., Keays, DA. (2012) Clusters of iron-rich cells in the upper beak of pigeons are macrophages not magnetosensitive neurons. Nature. 484(7394):367-70

2011

Edwards, A., Treiber, CD., Breuss, M., Pidsley, R., Huang, GJ., Cleak, J., Oliver, PL., Flint, J., Keays, DA. (2011) Cytoarchitectural disruption of the superior colliculus and an enlarged acoustic startle response in the Tuba1a mutant mouse. Neuroscience. 195:191-200
Stöger-Pollach, M., Treiber, CD., Resch, GP., Keays, DA., Ennen, I. (2011) EMCD real space maps of Magnetospirillum magnetotacticum. Micron. 42(5):456-60

2010

Braun, A., Breuss, M., Salzer, MC., Flint, J., Cowan, NJ., Keays, DA. (2010) Tuba8 is expressed at low levels in the developing mouse and human brain. Am J Hum Genet. 86(5):819-22; author reply 822-3
Keays, DA., Cleak, J., Huang, GJ., Edwards, A., Braun, A., Treiber, CD., Pidsley, R., Flint, J. (2010) The role of Tuba1a in adult hippocampal neurogenesis and the formation of the dentate gyrus. Dev Neurosci. 32(4):268-77
Tian, G., Jaglin, XH., Keays, DA., Francis, F., Chelly, J., Cowan, NJ. (2010) Disease-associated mutations in TUBA1A result in a spectrum of defects in the tubulin folding and heterodimer assembly pathway. Hum Mol Genet. 19(18):3599-613
Wincent, J., Bruno, DL., van Bon, BW., Bremer, A., Stewart, H., Bongers, EM., Ockeloen, CW., Willemsen, MH., Keays, DD., Baird, G., Newbury, DF., Kleefstra, T., Marcelis, C., Kini, U., Stark, Z., Savarirayan, R., Sheffield, LJ., Zuffardi, O., Slater, HR., de Vries, BB., Knight, SJ., Anderlid, BM., Schoumans, J. (2010) Sixteen New Cases Contributing to the Characterization of Patients with Distal 22q11.2 Microduplications. Mol Syndromol. 1(5):246-254

2009

Jaglin, XH., Poirier, K., Saillour, Y., Buhler, E., Tian, G., Bahi-Buisson, N., Fallet-Bianco, C., Phan-Dinh-Tuy, F., Kong, XP., Bomont, P., Castelnau-Ptakhine, L., Odent, S., Loget, P., Kossorotoff, M., Snoeck, I., Plessis, G., Parent, P., Beldjord, C., Cardoso, C., Represa, A., Flint, J., Keays, DA., Cowan, NJ., Chelly, J. (2009) Mutations in the beta-tubulin gene TUBB2B result in asymmetrical polymicrogyria. Nat Genet. 41(6):746-52

2008

Groszer, M., Keays, DA., Deacon, RM., de Bono, JP., Prasad-Mulcare, S., Gaub, S., Baum, MG., French, CA., Nicod, J., Coventry, JA., Enard, W., Fray, M., Brown, SD., Nolan, PM., Pääbo, S., Channon, KM., Costa, RM., Eilers, J., Ehret, G., Rawlins, JN., Fisher, SE. (2008) Impaired synaptic plasticity and motor learning in mice with a point mutation implicated in human speech deficits. Curr Biol. 18(5):354-62
Tian, G., Kong, XP., Jaglin, XH., Chelly, J., Keays, D., Cowan, NJ. (2008) A pachygyria-causing alpha-tubulin mutation results in inefficient cycling with CCT and a deficient interaction with TBCB. Mol Biol Cell. 19(3):1152-61

2007

Keays, DA., Clark, TG., Campbell, TG., Broxholme, J., Valdar, W. (2007) Estimating the number of coding mutations in genotypic and phenotypic driven N-ethyl-N-nitrosourea (ENU) screens: revisited. Mamm Genome. 18(2):123-4
Keays, DA., Tian, G., Poirier, K., Huang, GJ., Siebold, C., Cleak, J., Oliver, PL., Fray, M., Harvey, RJ., Molnár, Z., Piñon, MC., Dear, N., Valdar, W., Brown, SD., Davies, KE., Rawlins, JN., Cowan, NJ., Nolan, P., Chelly, J., Flint, J. (2007) Mutations in alpha-tubulin cause abnormal neuronal migration in mice and lissencephaly in humans. Cell. 128(1):45-57
Oliver, PL., Keays, DA., Davies, KE. (2007) Behavioural characterisation of the robotic mouse mutant. Behav Brain Res. 181(2):239-47
Poirier, K., Keays, DA., Francis, F., Saillour, Y., Bahi, N., Manouvrier, S., Fallet-Bianco, C., Pasquier, L., Toutain, A., Tuy, FP., Bienvenu, T., Joriot, S., Odent, S., Ville, D., Desguerre, I., Goldenberg, A., Moutard, ML., Fryns, JP., van Esch, H., Harvey, RJ., Siebold, C., Flint, J., Beldjord, C., Chelly, J. (2007) Large spectrum of lissencephaly and pachygyria phenotypes resulting from de novo missense mutations in tubulin alpha 1A (TUBA1A). Hum Mutat. 28(11):1055-64

2006

Livett, BG., Sandall, DW., Keays, D., Down, J., Gayler, KR., Satkunanathan, N., Khalil, Z. (2006) Therapeutic applications of conotoxins that target the neuronal nicotinic acetylcholine receptor. Toxicon. 48(7):810-29

2004

Bogani, D., Warr, N., Elms, P., Davies, J., Tymowska-Lalanne, Z., Goldsworthy, M., Cox, RD., Keays, DA., Flint, J., Wilson, V., Nolan, P., Arkell, R. (2004) New semidominant mutations that affect mouse development. Genesis. 40(2):109-117
Jakubowski, JA., Keays, DA., Kelley, WP., Sandall, DW., Bingham, JP., Livett, BG., Gayler, KR., Sweedler, JV. (2004) Determining sequences and post-translational modifications of novel conotoxins in Conus victoriae using cDNA sequencing and mass spectrometry. J Mass Spectrom. 39(5):548-57
Quwailid, MM., Hugill, A., Dear, N., Vizor, L., Wells, S., Horner, E., Fuller, S., Weedon, J., McMath, H., Woodman, P., Edwards, D., Campbell, D., Rodger, S., Carey, J., Roberts, A., Glenister, P., Lalanne, Z., Parkinson, N., Coghill, EL., McKeone, R., Cox, S., Willan, J., Greenfield, A., Keays, D., Brady, S., Spurr, N., Gray, I., Hunter, J., Brown, SD., Cox, RD. (2004) A gene-driven ENU-based approach to generating an allelic series in any gene. Mamm Genome. 15(8):585-91
Yalcin, B., Fullerton, J., Miller, S., Keays, DA., Brady, S., Bhomra, A., Jefferson, A., Volpi, E., Copley, RR., Flint, J., Mott, R. (2004) Unexpected complexity in the haplotypes of commonly used inbred strains of laboratory mice. Proc Natl Acad Sci U S A. 101(26):9734-9

2003

Keays, DA., Nolan, PM. (2003) N-ethyl-N-nitrosourea mouse mutants in the dissection of behavioural and psychiatric disorders. Eur J Pharmacol. 480(1-3):205-17
Sandall, DW., Satkunanathan, N., Keays, DA., Polidano, MA., Liping, X., Pham, V., Down, JG., Khalil, Z., Livett, BG., Gayler, KR. (2003) A novel alpha-conotoxin identified by gene sequencing is active in suppressing the vascular response to selective stimulation of sensory nerves in vivo. Biochemistry. 42(22):6904-11