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Utilizing microphysiological systems and induced pluripotent stem cells for disease modeling: a case study for blood brain barrier research in a pharmaceutical setting.

Fabre KM, Delsing L, Hicks R, Colclough N, Crowther DC, Ewart L.

Adv Drug Deliv Rev. 2019 Feb 1;140:129-135. doi: 10.1016/j.addr.2018.09.009. Epub 2018 Sep 22. Review.


Prion protein stabilizes amyloid-β (Aβ) oligomers and enhances Aβ neurotoxicity in a Drosophila model of Alzheimer's disease.

Younan ND, Chen KF, Rose RS, Crowther DC, Viles JH.

J Biol Chem. 2018 Aug 24;293(34):13090-13099. doi: 10.1074/jbc.RA118.003319. Epub 2018 Jun 10.


Central and peripheral circadian clocks and their role in Alzheimer's disease.

Chauhan R, Chen KF, Kent BA, Crowther DC.

Dis Model Mech. 2017 Oct 1;10(10):1187-1199. doi: 10.1242/dmm.030627. Review.


TREM2 shedding by cleavage at the H157-S158 bond is accelerated for the Alzheimer's disease-associated H157Y variant.

Thornton P, Sevalle J, Deery MJ, Fraser G, Zhou Y, Ståhl S, Franssen EH, Dodd RB, Qamar S, Gomez Perez-Nievas B, Nicol LS, Eketjäll S, Revell J, Jones C, Billinton A, St George-Hyslop PH, Chessell I, Crowther DC.

EMBO Mol Med. 2017 Oct;9(10):1366-1378. doi: 10.15252/emmm.201707673.


CCT complex restricts neuropathogenic protein aggregation via autophagy.

Pavel M, Imarisio S, Menzies FM, Jimenez-Sanchez M, Siddiqi FH, Wu X, Renna M, O'Kane CJ, Crowther DC, Rubinsztein DC.

Nat Commun. 2016 Dec 8;7:13821. doi: 10.1038/ncomms13821.


Flyglow: Single-fly observations of simultaneous molecular and behavioural circadian oscillations in controls and an Alzheimer's model.

Khabirova E, Chen KF, O'Neill JS, Crowther DC.

Sci Rep. 2016 Sep 23;6:33759. doi: 10.1038/srep33759.


Progressive myoclonus epilepsy associated with neuroserpin inclusion bodies (neuroserpinosis).

Roussel BD, Lomas DA, Crowther DC.

Epileptic Disord. 2016 Sep 1;18(S2):103-110. Review.


Metabolic changes may precede proteostatic dysfunction in a Drosophila model of amyloid beta peptide toxicity.

Ott S, Vishnivetskaya A, Malmendal A, Crowther DC.

Neurobiol Aging. 2016 May;41:39-52. doi: 10.1016/j.neurobiolaging.2016.01.009. Epub 2016 Feb 11.


Expression of the alternative oxidase mitigates beta-amyloid production and toxicity in model systems.

El-Khoury R, Kaulio E, Lassila KA, Crowther DC, Jacobs HT, Rustin P.

Free Radic Biol Med. 2016 Jul;96:57-66. doi: 10.1016/j.freeradbiomed.2016.04.006. Epub 2016 Apr 14.


Drosophila melanogaster as a Model for Studies on the Early Stages of Alzheimer's Disease.

Lim JY, Ott S, Crowther DC.

Methods Mol Biol. 2016;1303:227-39. doi: 10.1007/978-1-4939-2627-5_13.


Iron is a specific cofactor for distinct oxidation- and aggregation-dependent Aβ toxicity mechanisms in a Drosophila model.

Ott S, Dziadulewicz N, Crowther DC.

Dis Model Mech. 2015 Jul 1;8(7):657-67. doi: 10.1242/dmm.019042. Epub 2015 Apr 23.


The pro-domains of neurotrophins, including BDNF, are linked to Alzheimer's disease through a toxic synergy with Aβ.

Lim JY, Reighard CP, Crowther DC.

Hum Mol Genet. 2015 Jul 15;24(14):3929-38. doi: 10.1093/hmg/ddv130. Epub 2015 May 7.


Insights into amyloid disease from fly models.

Chen KF, Crowther DC.

Essays Biochem. 2014;56:69-83. doi: 10.1042/bse0560069.


The TRiC/CCT chaperone is implicated in Alzheimer's disease based on patient GWAS and an RNAi screen in Aβ-expressing Caenorhabditis elegans.

Khabirova E, Moloney A, Marciniak SJ, Williams J, Lomas DA, Oliver SG, Favrin G, Sattelle DB, Crowther DC.

PLoS One. 2014 Jul 31;9(7):e102985. doi: 10.1371/journal.pone.0102985. eCollection 2014.


Oligomer-targeting with a conformational antibody fragment promotes toxicity in Aβ-expressing flies.

Wacker J, Rönicke R, Westermann M, Wulff M, Reymann KG, Dobson CM, Horn U, Crowther DC, Luheshi LM, Fändrich M.

Acta Neuropathol Commun. 2014 Apr 11;2:43. doi: 10.1186/2051-5960-2-43.


Retro-inversal of intracellular selected β-amyloid-interacting peptides: implications for a novel Alzheimer's disease treatment.

Acerra N, Kad NM, Griffith DA, Ott S, Crowther DC, Mason JM.

Biochemistry. 2014 Apr 8;53(13):2101-11. doi: 10.1021/bi5001257. Epub 2014 Mar 21.


The central molecular clock is robust in the face of behavioural arrhythmia in a Drosophila model of Alzheimer's disease.

Chen KF, Possidente B, Lomas DA, Crowther DC.

Dis Model Mech. 2014 Apr;7(4):445-58. doi: 10.1242/dmm.014134. Epub 2014 Feb 26.


Identification of novel modifiers of Aβ toxicity by transcriptomic analysis in the fruitfly.

Favrin G, Bean DM, Bilsland E, Boyer H, Fischer BE, Russell S, Crowther DC, Baylis HA, Oliver SG, Giannakou ME.

Sci Rep. 2013 Dec 16;3:3512. doi: 10.1038/srep03512.


Suppression of Aβ toxicity by puromycin-sensitive aminopeptidase is independent of its proteolytic activity.

Kruppa AJ, Ott S, Chandraratna DS, Irving JA, Page RM, Speretta E, Seto T, Camargo LM, Marciniak SJ, Lomas DA, Crowther DC.

Biochim Biophys Acta. 2013 Dec;1832(12):2115-26. doi: 10.1016/j.bbadis.2013.07.019. Epub 2013 Aug 2.


Sterol metabolism regulates neuroserpin polymer degradation in the absence of the unfolded protein response in the dementia FENIB.

Roussel BD, Newton TM, Malzer E, Simecek N, Haq I, Thomas SE, Burr ML, Lehner PJ, Crowther DC, Marciniak SJ, Lomas DA.

Hum Mol Genet. 2013 Nov 15;22(22):4616-26. doi: 10.1093/hmg/ddt310. Epub 2013 Jun 28.


Coordinate regulation of eIF2α phosphorylation by PPP1R15 and GCN2 is required during Drosophila development.

Malzer E, Szajewska-Skuta M, Dalton LE, Thomas SE, Hu N, Skaer H, Lomas DA, Crowther DC, Marciniak SJ.

J Cell Sci. 2013 Mar 15;126(Pt 6):1406-15. doi: 10.1242/jcs.117614. Epub 2013 Feb 15.


p53 and translation attenuation regulate distinct cell cycle checkpoints during endoplasmic reticulum (ER) stress.

Thomas SE, Malzer E, Ordóñez A, Dalton LE, van 't Wout EF, Liniker E, Crowther DC, Lomas DA, Marciniak SJ.

J Biol Chem. 2013 Mar 15;288(11):7606-17. doi: 10.1074/jbc.M112.424655. Epub 2013 Jan 22.


Endoplasmic reticulum dysfunction in neurological disease.

Roussel BD, Kruppa AJ, Miranda E, Crowther DC, Lomas DA, Marciniak SJ.

Lancet Neurol. 2013 Jan;12(1):105-18. doi: 10.1016/S1474-4422(12)70238-7. Review.


Functional genomics in Drosophila models of human disease.

Chen KF, Crowther DC.

Brief Funct Genomics. 2012 Sep;11(5):405-15. doi: 10.1093/bfgp/els038. Epub 2012 Aug 22. Review.


Expression in drosophila of tandem amyloid β peptides provides insights into links between aggregation and neurotoxicity.

Speretta E, Jahn TR, Tartaglia GG, Favrin G, Barros TP, Imarisio S, Lomas DA, Luheshi LM, Crowther DC, Dobson CM.

J Biol Chem. 2012 Jun 8;287(24):20748-54. doi: 10.1074/jbc.M112.350124. Epub 2012 Mar 29.


Ovine PrP transgenic Drosophila show reduced locomotor activity and decreased survival.

Thackray AM, Muhammad F, Zhang C, Di Y, Jahn TR, Landgraf M, Crowther DC, Evers JF, Bujdoso R.

Biochem J. 2012 Jun 15;444(3):487-95. doi: 10.1042/BJ20112141.


Prion-induced toxicity in PrP transgenic Drosophila.

Thackray AM, Muhammad F, Zhang C, Denyer M, Spiropoulos J, Crowther DC, Bujdoso R.

Exp Mol Pathol. 2012 Apr;92(2):194-201. doi: 10.1016/j.yexmp.2012.01.005. Epub 2012 Jan 31.


Testing the therapeutic potential of doxycycline in a Drosophila melanogaster model of Alzheimer disease.

Costa R, Speretta E, Crowther DC, Cardoso I.

J Biol Chem. 2011 Dec 2;286(48):41647-55. doi: 10.1074/jbc.M111.274548. Epub 2011 Oct 13.


Disease-related amyloidogenic variants of human lysozyme trigger the unfolded protein response and disturb eye development in Drosophila melanogaster.

Kumita JR, Helmfors L, Williams J, Luheshi LM, Menzer L, Dumoulin M, Lomas DA, Crowther DC, Dobson CM, Brorsson AC.

FASEB J. 2012 Jan;26(1):192-202. doi: 10.1096/fj.11-185983. Epub 2011 Sep 29.


The iFly tracking system for an automated locomotor and behavioural analysis of Drosophila melanogaster.

Kohlhoff KJ, Jahn TR, Lomas DA, Dobson CM, Crowther DC, Vendruscolo M.

Integr Biol (Camb). 2011 Jul;3(7):755-60. doi: 10.1039/c0ib00149j. Epub 2011 Jun 23.


Modeling serpin conformational diseases in Drosophila melanogaster.

Jahn TR, Malzer E, Roote J, Vishnivetskaya A, Imarisio S, Giannakou M, Panser K, Marciniak S, Crowther DC.

Methods Enzymol. 2011;499:227-58. doi: 10.1016/B978-0-12-386471-0.00012-2.


Unravelling the twists and turns of the serpinopathies.

Roussel BD, Irving JA, Ekeowa UI, Belorgey D, Haq I, Ordóñez A, Kruppa AJ, Duvoix A, Rashid ST, Crowther DC, Marciniak SJ, Lomas DA.

FEBS J. 2011 Oct;278(20):3859-67. doi: 10.1111/j.1742-4658.2011.08201.x. Epub 2011 Jun 20. Review.


Detection of early locomotor abnormalities in a Drosophila model of Alzheimer's disease.

Jahn TR, Kohlhoff KJ, Scott M, Tartaglia GG, Lomas DA, Dobson CM, Vendruscolo M, Crowther DC.

J Neurosci Methods. 2011 Apr 15;197(1):186-9. doi: 10.1016/j.jneumeth.2011.01.026. Epub 2011 Feb 18.


Iron promotes the toxicity of amyloid beta peptide by impeding its ordered aggregation.

Liu B, Moloney A, Meehan S, Morris K, Thomas SE, Serpell LC, Hider R, Marciniak SJ, Lomas DA, Crowther DC.

J Biol Chem. 2011 Feb 11;286(6):4248-56. doi: 10.1074/jbc.M110.158980. Epub 2010 Dec 8.


Characterisation of serpin polymers in vitro and in vivo.

Belorgey D, Irving JA, Ekeowa UI, Freeke J, Roussel BD, Miranda E, Pérez J, Robinson CV, Marciniak SJ, Crowther DC, Michel CH, Lomas DA.

Methods. 2011 Mar;53(3):255-66. doi: 10.1016/j.ymeth.2010.11.008. Epub 2010 Nov 27. Review.


Impaired tissue growth is mediated by checkpoint kinase 1 (CHK1) in the integrated stress response.

Malzer E, Daly ML, Moloney A, Sendall TJ, Thomas SE, Ryder E, Ryoo HD, Crowther DC, Lomas DA, Marciniak SJ.

J Cell Sci. 2010 Sep 1;123(Pt 17):2892-900. doi: 10.1242/jcs.070078. Epub 2010 Aug 3.


ANS binding reveals common features of cytotoxic amyloid species.

Bolognesi B, Kumita JR, Barros TP, Esbjorner EK, Luheshi LM, Crowther DC, Wilson MR, Dobson CM, Favrin G, Yerbury JJ.

ACS Chem Biol. 2010 Aug 20;5(8):735-40. doi: 10.1021/cb1001203.


Intrinsic determinants of neurotoxic aggregate formation by the amyloid beta peptide.

Brorsson AC, Bolognesi B, Tartaglia GG, Shammas SL, Favrin G, Watson I, Lomas DA, Chiti F, Vendruscolo M, Dobson CM, Crowther DC, Luheshi LM.

Biophys J. 2010 Apr 21;98(8):1677-84. doi: 10.1016/j.bpj.2009.12.4320.


Sequestration of the Abeta peptide prevents toxicity and promotes degradation in vivo.

Luheshi LM, Hoyer W, de Barros TP, van Dijk Härd I, Brorsson AC, Macao B, Persson C, Crowther DC, Lomas DA, Ståhl S, Dobson CM, Härd T.

PLoS Biol. 2010 Mar 16;8(3):e1000334. doi: 10.1371/journal.pbio.1000334.


Methods and models in neurodegenerative and systemic protein aggregation diseases.

Brorsson AC, Kumita JR, MacLeod I, Bolognesi B, Speretta E, Luheshi LM, Knowles TP, Dobson CM, Crowther DC.

Front Biosci (Landmark Ed). 2010 Jan 1;15:373-96. Review.


Alzheimer's disease: insights from Drosophila melanogaster models.

Moloney A, Sattelle DB, Lomas DA, Crowther DC.

Trends Biochem Sci. 2010 Apr;35(4):228-35. doi: 10.1016/j.tibs.2009.11.004. Epub 2009 Dec 25. Review.


Endoplasmic reticulum-associated degradation (ERAD) and autophagy cooperate to degrade polymerogenic mutant serpins.

Kroeger H, Miranda E, MacLeod I, Pérez J, Crowther DC, Marciniak SJ, Lomas DA.

J Biol Chem. 2009 Aug 21;284(34):22793-802. doi: 10.1074/jbc.M109.027102. Epub 2009 Jun 23.


Fenton chemistry and oxidative stress mediate the toxicity of the beta-amyloid peptide in a Drosophila model of Alzheimer's disease.

Rival T, Page RM, Chandraratna DS, Sendall TJ, Ryder E, Liu B, Lewis H, Rosahl T, Hider R, Camargo LM, Shearman MS, Crowther DC, Lomas DA.

Eur J Neurosci. 2009 Apr;29(7):1335-47. doi: 10.1111/j.1460-9568.2009.06701.x. Epub 2009 Mar 23.


Alpha-helix targeting reduces amyloid-beta peptide toxicity.

Nerelius C, Sandegren A, Sargsyan H, Raunak R, Leijonmarck H, Chatterjee U, Fisahn A, Imarisio S, Lomas DA, Crowther DC, Strömberg R, Johansson J.

Proc Natl Acad Sci U S A. 2009 Jun 9;106(23):9191-6. doi: 10.1073/pnas.0810364106. Epub 2009 May 20.


alpha1-Antitrypsin deficiency, chronic obstructive pulmonary disease and the serpinopathies.

Ekeowa UI, Gooptu B, Belorgey D, Hägglöf P, Karlsson-Li S, Miranda E, Pérez J, MacLeod I, Kroger H, Marciniak SJ, Crowther DC, Lomas DA.

Clin Sci (Lond). 2009 May 14;116(12):837-50. doi: 10.1042/CS20080484. Review.


Using a Drosophila model of Alzheimer's disease.

Crowther DC, Page R, Rival T, Chandraratna DS, Lomas DA.

SEB Exp Biol Ser. 2008;60:57-77. Review. No abstract available.


Protein misfolding and disease: from the test tube to the organism.

Luheshi LM, Crowther DC, Dobson CM.

Curr Opin Chem Biol. 2008 Feb;12(1):25-31. doi: 10.1016/j.cbpa.2008.02.011. Epub 2008 Mar 18. Review.


The intracellular accumulation of polymeric neuroserpin explains the severity of the dementia FENIB.

Miranda E, MacLeod I, Davies MJ, Pérez J, Römisch K, Crowther DC, Lomas DA.

Hum Mol Genet. 2008 Jun 1;17(11):1527-39. doi: 10.1093/hmg/ddn041. Epub 2008 Feb 11.


Systematic in vivo analysis of the intrinsic determinants of amyloid Beta pathogenicity.

Luheshi LM, Tartaglia GG, Brorsson AC, Pawar AP, Watson IE, Chiti F, Vendruscolo M, Lomas DA, Dobson CM, Crowther DC.

PLoS Biol. 2007 Oct 30;5(11):e290.


Plasma and CSF serpins in Alzheimer disease and dementia with Lewy bodies.

Nielsen HM, Minthon L, Londos E, Blennow K, Miranda E, Perez J, Crowther DC, Lomas DA, Janciauskiene SM.

Neurology. 2007 Oct 16;69(16):1569-79. Epub 2007 Aug 29.


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