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What is the level of dystrophin expression required for effective therapy of Duchenne muscular dystrophy?

Wells DJ.

J Muscle Res Cell Motil. 2019 Jun;40(2):141-150. doi: 10.1007/s10974-019-09535-9. Epub 2019 Jul 9.


Identification of qPCR reference genes suitable for normalizing gene expression in the mdx mouse model of Duchenne muscular dystrophy.

Hildyard JCW, Finch AM, Wells DJ.

PLoS One. 2019 Jan 30;14(1):e0211384. doi: 10.1371/journal.pone.0211384. eCollection 2019.


Cmah-dystrophin deficient mdx mice display an accelerated cardiac phenotype that is improved following peptide-PMO exon skipping treatment.

Betts CA, McClorey G, Healicon R, Hammond SM, Manzano R, Muses S, Ball V, Godfrey C, Merritt TM, van Westering T, O'Donovan L, Wells KE, Gait MJ, Wells DJ, Tyler D, Wood MJ.

Hum Mol Genet. 2019 Feb 1;28(3):396-406. doi: 10.1093/hmg/ddy346.


"Of Mice and Measures": A Project to Improve How We Advance Duchenne Muscular Dystrophy Therapies to the Clinic.

Gordish-Dressman H, Willmann R, Dalle Pazze L, Kreibich A, van Putten M, Heydemann A, Bogdanik L, Lutz C, Davies K, Demonbreun AR, Duan D, Elsey D, Fukada SI, Girgenrath M, Patrick Gonzalez J, Grounds MD, Nichols A, Partridge T, Passini M, Sanarica F, Schnell FJ, Wells DJ, Yokota T, Young CS, Zhong Z, Spurney C, Spencer M, De Luca A, Nagaraju K, Aartsma-Rus A.

J Neuromuscul Dis. 2018;5(4):407-417. doi: 10.3233/JND-180324.


Tracking progress: an update on animal models for Duchenne muscular dystrophy.

Wells DJ.

Dis Model Mech. 2018 Jun 13;11(6). pii: dmm035774. doi: 10.1242/dmm.035774.


Three-Dimensional Human iPSC-Derived Artificial Skeletal Muscles Model Muscular Dystrophies and Enable Multilineage Tissue Engineering.

Maffioletti SM, Sarcar S, Henderson ABH, Mannhardt I, Pinton L, Moyle LA, Steele-Stallard H, Cappellari O, Wells KE, Ferrari G, Mitchell JS, Tyzack GE, Kotiadis VN, Khedr M, Ragazzi M, Wang W, Duchen MR, Patani R, Zammit PS, Wells DJ, Eschenhagen T, Tedesco FS.

Cell Rep. 2018 Apr 17;23(3):899-908. doi: 10.1016/j.celrep.2018.03.091.


Determination of qPCR Reference Genes Suitable for Normalizing Gene Expression in a Canine Model of Duchenne Muscular Dystrophy.

Hildyard JCW, Taylor-Brown F, Massey C, Wells DJ, Piercy RJ.

J Neuromuscul Dis. 2018;5(2):177-191. doi: 10.3233/JND-170267.


The inter-tester repeatability of a model for analysing elbow flexion-extension during overhead sporting movements.

Wells DJM, Donnelly CJ, Elliott BC, Middleton KJ, Alderson JA.

Med Biol Eng Comput. 2018 Oct;56(10):1853-1860. doi: 10.1007/s11517-018-1820-5. Epub 2018 Apr 3.


Focus on the Role of D-serine and D-amino Acid Oxidase in Amyotrophic Lateral Sclerosis/Motor Neuron Disease (ALS).

Kondori NR, Paul P, Robbins JP, Liu K, Hildyard JCW, Wells DJ, de Belleroche JS.

Front Mol Biosci. 2018 Feb 13;5:8. doi: 10.3389/fmolb.2018.00008. eCollection 2018. Review.


Characterisation of the pathogenic effects of the in vivo expression of an ALS-linked mutation in D-amino acid oxidase: Phenotype and loss of spinal cord motor neurons.

Kondori NR, Paul P, Robbins JP, Liu K, Hildyard JCW, Wells DJ, de Belleroche JS.

PLoS One. 2017 Dec 1;12(12):e0188912. doi: 10.1371/journal.pone.0188912. eCollection 2017.


Designing translationally relevant preclinical studies of new therapeutics.

Wells DJ.

Exp Physiol. 2017 Jun 1;102(6):616. doi: 10.1113/EP086296. Epub 2017 May 14. No abstract available.


Systemic AAV Gene Therapy Close to Clinical Trials for Several Neuromuscular Diseases.

Wells DJ.

Mol Ther. 2017 Apr 5;25(4):834-835. doi: 10.1016/j.ymthe.2017.03.006. Epub 2017 Mar 22. No abstract available.


Prescribing joint co-ordinates during model preparation to improve inverse kinematic estimates of elbow joint angles.

Wells DJ, Alderson JA, Dunne J, Elliott BC, Donnelly CJ.

J Biomech. 2017 Jan 25;51:111-117. doi: 10.1016/j.jbiomech.2016.11.057. Epub 2016 Nov 29.


Histopathological Evaluation of Skeletal Muscle with Specific Reference to Mouse Models of Muscular Dystrophy.

Terry RL, Wells DJ.

Curr Protoc Mouse Biol. 2016 Dec 1;6(4):343-363. doi: 10.1002/cpmo.19.


Investigating Synthetic Oligonucleotide Targeting of Mir31 in Duchenne Muscular Dystrophy.

Hildyard JC, Wells DJ.

PLoS Curr. 2016 Jun 16;8. pii: doi: 10.1371/


Transgenic Rescue of the LARGEmyd Mouse: A LARGE Therapeutic Window?

Hildyard JC, Lacey E, Booler H, Hopkinson M, Wells DJ, Brown SC.

PLoS One. 2016 Jul 28;11(7):e0159853. doi: 10.1371/journal.pone.0159853. eCollection 2016.


Growth differentiation factor-15 is associated with muscle mass in chronic obstructive pulmonary disease and promotes muscle wasting in vivo.

Patel MS, Lee J, Baz M, Wells CE, Bloch S, Lewis A, Donaldson AV, Garfield BE, Hopkinson NS, Natanek A, Man WD, Wells DJ, Baker EH, Polkey MI, Kemp PR.

J Cachexia Sarcopenia Muscle. 2016 Sep;7(4):436-48. doi: 10.1002/jcsm.12096. Epub 2015 Dec 29.


Muscle moment arms and sensitivity analysis of a mouse hindlimb musculoskeletal model.

Charles JP, Cappellari O, Spence AJ, Wells DJ, Hutchinson JR.

J Anat. 2016 Oct;229(4):514-35. doi: 10.1111/joa.12461. Epub 2016 May 12.


Musculoskeletal Geometry, Muscle Architecture and Functional Specialisations of the Mouse Hindlimb.

Charles JP, Cappellari O, Spence AJ, Hutchinson JR, Wells DJ.

PLoS One. 2016 Apr 26;11(4):e0147669. doi: 10.1371/journal.pone.0147669. eCollection 2016.


Olfaction variation in mouse husbandry and its implications for refinement and standardization: UK survey of animal scents.

López-Salesansky N, Mazlan NH, Whitfield LE, Wells DJ, Burn CC.

Lab Anim. 2016 Oct;50(5):362-9. doi: 10.1177/0023677215622883. Epub 2015 Dec 20.


Olfactory variation in mouse husbandry and its implications for refinement and standardization: UK survey of non-animal scents.

López-Salesansky N, Mazlan NH, Whitfield LE, Wells DJ, Burn CC.

Lab Anim. 2016 Aug;50(4):286-95. doi: 10.1177/0023677215614296. Epub 2015 Nov 10.


FHL1 activates myostatin signalling in skeletal muscle and promotes atrophy.

Lee JY, Lori D, Wells DJ, Kemp PR.

FEBS Open Bio. 2015 Sep 1;5:753-62. doi: 10.1016/j.fob.2015.08.011. eCollection 2015.


Implications for Cardiac Function Following Rescue of the Dystrophic Diaphragm in a Mouse Model of Duchenne Muscular Dystrophy.

Betts CA, Saleh AF, Carr CA, Muses S, Wells KE, Hammond SM, Godfrey C, McClorey G, Woffindale C, Clarke K, Wells DJ, Gait MJ, Wood MJ.

Sci Rep. 2015 Jun 26;5:11632. doi: 10.1038/srep11632.


How much dystrophin is enough: the physiological consequences of different levels of dystrophin in the mdx mouse.

Godfrey C, Muses S, McClorey G, Wells KE, Coursindel T, Terry RL, Betts C, Hammond S, O'Donovan L, Hildyard J, El Andaloussi S, Gait MJ, Wood MJ, Wells DJ.

Hum Mol Genet. 2015 Aug 1;24(15):4225-37. doi: 10.1093/hmg/ddv155. Epub 2015 May 1.


The TREAT-NMD advisory committee for therapeutics (TACT): an innovative de-risking model to foster orphan drug development.

Heslop E, Csimma C, Straub V, McCall J, Nagaraju K, Wagner KR, Caizergues D, Korinthenberg R, Flanigan KM, Kaufmann P, McNeil E, Mendell J, Hesterlee S, Wells DJ, Bushby K; TACT.

Orphanet J Rare Dis. 2015 Apr 23;10:49. doi: 10.1186/s13023-015-0258-1. Review.


Measuring clinical effectiveness of medicinal products for the treatment of Duchenne muscular dystrophy.

Lynn S, Aartsma-Rus A, Bushby K, Furlong P, Goemans N, De Luca A, Mayhew A, McDonald C, Mercuri E, Muntoni F, Pohlschmidt M, Verschuuren J, Voit T, Vroom E, Wells DJ, Straub V.

Neuromuscul Disord. 2015 Jan;25(1):96-105. doi: 10.1016/j.nmd.2014.09.003. Epub 2014 Sep 11. No abstract available.


Translational and regulatory challenges for exon skipping therapies.

Aartsma-Rus A, Ferlini A, Goemans N, Pasmooij AM, Wells DJ, Bushby K, Vroom E, Balabanov P.

Hum Gene Ther. 2014 Oct;25(10):885-92. doi: 10.1089/hum.2014.086. Review.


Poloxamer [corrected] 188 has a deleterious effect on dystrophic skeletal muscle function.

Terry RL, Kaneb HM, Wells DJ.

PLoS One. 2014 Mar 18;9(3):e91221. doi: 10.1371/journal.pone.0091221. eCollection 2014. Erratum in: PLoS One. 2015;10(3):e0119252.


Identification and validation of quantitative PCR reference genes suitable for normalizing expression in normal and dystrophic cell culture models of myogenesis.

Hildyard JC, Wells DJ.

PLoS Curr. 2014 Mar 6;6. pii: doi: 10.1371/


The transgenic expression of LARGE exacerbates the muscle phenotype of dystroglycanopathy mice.

Whitmore C, Fernandez-Fuente M, Booler H, Parr C, Kavishwar M, Ashraf A, Lacey E, Kim J, Terry R, Ackroyd MR, Wells KE, Muntoni F, Wells DJ, Brown SC.

Hum Mol Genet. 2014 Apr 1;23(7):1842-55. doi: 10.1093/hmg/ddt577. Epub 2013 Nov 13.


Preventing phosphorylation of dystroglycan ameliorates the dystrophic phenotype in mdx mouse.

Miller G, Moore CJ, Terry R, La Riviere T, Mitchell A, Piggott R, Dear TN, Wells DJ, Winder SJ.

Hum Mol Genet. 2012 Oct 15;21(20):4508-20. Epub 2012 Jul 18.


Effect of albumin and dextrose concentration on ultrasound and microbubble mediated gene transfection in vivo.

Browning RJ, Mulvana H, Tang MX, Hajnal JV, Wells DJ, Eckersley RJ.

Ultrasound Med Biol. 2012 Jun;38(6):1067-77. doi: 10.1016/j.ultrasmedbio.2012.02.020. Epub 2012 Apr 12.


Restoration of dystrophin expression using the Sleeping Beauty transposon.

Muses S, Morgan JE, Wells DJ.

Version 2. PLoS Curr. 2011 Dec 22 [revised 2012 Jan 1];3:RRN1296.


Animal welfare and the 3Rs in European biomedical research.

Wells DJ.

Ann N Y Acad Sci. 2011 Dec;1245:14-6. doi: 10.1111/j.1749-6632.2011.06335.x.


Förster resonance energy transfer imaging in vivo with approximated radiative transfer equation.

Soloviev VY, McGinty J, Stuckey DW, Laine R, Wylezinska-Arridge M, Wells DJ, Sardini A, Hajnal JV, French PM, Arridge SR.

Appl Opt. 2011 Dec 20;50(36):6583-90. doi: 10.1364/AO.50.006583.


A new extensively characterised conditionally immortal muscle cell-line for investigating therapeutic strategies in muscular dystrophies.

Muses S, Morgan JE, Wells DJ.

PLoS One. 2011;6(9):e24826. doi: 10.1371/journal.pone.0024826. Epub 2011 Sep 14.


Metformin treatment has no beneficial effect in a dose-response survival study in the SOD1(G93A) mouse model of ALS and is harmful in female mice.

Kaneb HM, Sharp PS, Rahmani-Kondori N, Wells DJ.

PLoS One. 2011;6(9):e24189. doi: 10.1371/journal.pone.0024189. Epub 2011 Sep 1.


Muscular dystrophies due to glycosylation defects: diagnosis and therapeutic strategies.

Muntoni F, Torelli S, Wells DJ, Brown SC.

Curr Opin Neurol. 2011 Oct;24(5):437-42. doi: 10.1097/WCO.0b013e32834a95e3. Review.


Exon skipping and dystrophin restoration in patients with Duchenne muscular dystrophy after systemic phosphorodiamidate morpholino oligomer treatment: an open-label, phase 2, dose-escalation study.

Cirak S, Arechavala-Gomeza V, Guglieri M, Feng L, Torelli S, Anthony K, Abbs S, Garralda ME, Bourke J, Wells DJ, Dickson G, Wood MJ, Wilton SD, Straub V, Kole R, Shrewsbury SB, Sewry C, Morgan JE, Bushby K, Muntoni F.

Lancet. 2011 Aug 13;378(9791):595-605. doi: 10.1016/S0140-6736(11)60756-3. Epub 2011 Jul 23.


In vivo fluorescence lifetime tomography of a FRET probe expressed in mouse.

McGinty J, Stuckey DW, Soloviev VY, Laine R, Wylezinska-Arridge M, Wells DJ, Arridge SR, French PM, Hajnal JV, Sardini A.

Biomed Opt Express. 2011 Jul 1;2(7):1907-17. doi: 10.1364/BOE.2.001907. Epub 2011 Jun 10.


Influence of needle gauge on in vivo ultrasound and microbubble-mediated gene transfection.

Browning RJ, Mulvana H, Tang M, Hajnal JV, Wells DJ, Eckersley RJ.

Ultrasound Med Biol. 2011 Sep;37(9):1531-7. doi: 10.1016/j.ultrasmedbio.2011.05.019. Epub 2011 Jul 7.


Transgenic overexpression of LARGE induces α-dystroglycan hyperglycosylation in skeletal and cardiac muscle.

Brockington M, Torelli S, Sharp PS, Liu K, Cirak S, Brown SC, Wells DJ, Muntoni F.

PLoS One. 2010 Dec 28;5(12):e14434. doi: 10.1371/journal.pone.0014434.


Chronic systemic therapy with low-dose morpholino oligomers ameliorates the pathology and normalizes locomotor behavior in mdx mice.

Malerba A, Sharp PS, Graham IR, Arechavala-Gomeza V, Foster K, Muntoni F, Wells DJ, Dickson G.

Mol Ther. 2011 Feb;19(2):345-54. doi: 10.1038/mt.2010.261. Epub 2010 Nov 23.


Physiological characterization of muscle strength with variable levels of dystrophin restoration in mdx mice following local antisense therapy.

Sharp PS, Bye-a-Jee H, Wells DJ.

Mol Ther. 2011 Jan;19(1):165-71. doi: 10.1038/mt.2010.213. Epub 2010 Oct 5.


Coping in men with prostate cancer and their partners: a quantitative and qualitative study.

Green HJ, Wells DJ, Laakso L.

Eur J Cancer Care (Engl). 2011 Mar;20(2):237-47. doi: 10.1111/j.1365-2354.2010.01225.x. Epub 2010 Sep 6.


Genetically modified animals and pharmacological research.

Wells DJ.

Handb Exp Pharmacol. 2010;(199):213-26. doi: 10.1007/978-3-642-10324-7_9. Review.


Overexpression of heat shock protein 27 reduces cortical damage after cerebral ischemia.

van der Weerd L, Tariq Akbar M, Aron Badin R, Valentim LM, Thomas DL, Wells DJ, Latchman DS, Gadian DG, Lythgoe MF, de Belleroche JS.

J Cereb Blood Flow Metab. 2010 Apr;30(4):849-56. doi: 10.1038/jcbfm.2009.249. Epub 2009 Dec 9.


Electroporation and ultrasound enhanced non-viral gene delivery in vitro and in vivo.

Wells DJ.

Cell Biol Toxicol. 2010 Feb;26(1):21-8. doi: 10.1007/s10565-009-9144-8. Epub 2009 Dec 1. Review.


Heat shock protein 27 protects against atherogenesis via an estrogen-dependent mechanism: role of selective estrogen receptor beta modulation.

Rayner K, Sun J, Chen YX, McNulty M, Simard T, Zhao X, Wells DJ, de Belleroche J, O'Brien ER.

Arterioscler Thromb Vasc Biol. 2009 Nov;29(11):1751-6. doi: 10.1161/ATVBAHA.109.193656. Epub 2009 Sep 3.


Local restoration of dystrophin expression with the morpholino oligomer AVI-4658 in Duchenne muscular dystrophy: a single-blind, placebo-controlled, dose-escalation, proof-of-concept study.

Kinali M, Arechavala-Gomeza V, Feng L, Cirak S, Hunt D, Adkin C, Guglieri M, Ashton E, Abbs S, Nihoyannopoulos P, Garralda ME, Rutherford M, McCulley C, Popplewell L, Graham IR, Dickson G, Wood MJ, Wells DJ, Wilton SD, Kole R, Straub V, Bushby K, Sewry C, Morgan JE, Muntoni F.

Lancet Neurol. 2009 Oct;8(10):918-28. doi: 10.1016/S1474-4422(09)70211-X. Epub 2009 Aug 25. Erratum in: Lancet Neurol. 2009 Dec;8(12):1083.

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