Research Topics
Species | T M CoxSummaryAffiliation: University of Cambridge Country: UK Publications
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Publications
Alkaptonuria: leading to the treasure in exceptionsTimothy M Cox
University of Cambridge, Cambridge, UK
JIMD Rep 5:49-57. 2012....
The Cambridge Bachelor of Medicine (MB)/Doctor of Philosophy (PhD): graduate outcomes of the first MB/PhD programme in the UKTimothy M Cox
General Practice and Primary Care Research Unit, Department of Public Health and Primary Care Institute of Public Health, Cambridge
Clin Med 12:530-4. 2012....
Evaluation of miglustat as maintenance therapy after enzyme therapy in adults with stable type 1 Gaucher disease: a prospective, open-label non-inferiority studyTimothy M Cox
University of Cambridge, Addenbrooke s Hospital, Cambridge, UK
Orphanet J Rare Dis 7:102. 2012..abstract:..
Management of non-neuronopathic Gaucher disease with special reference to pregnancy, splenectomy, bisphosphonate therapy, use of biomarkers and bone disease monitoringT M Cox
Department of Medicine, University of Cambridge, Addenbrooke s NHS Foundation Hospitals Trust, Cambridge, UK
J Inherit Metab Dis 31:319-36. 2008....
Eliglustat tartrate, an orally active glucocerebroside synthase inhibitor for the potential treatment of Gaucher disease and other lysosomal storage diseasesTimothy M Cox
University of Cambridge, Department of Medicine, Addenbrooke s Hospital, Cambridge, UK
Curr Opin Investig Drugs 11:1169-81. 2010..Eliglustat tartrate is orally active and, with potent effects on the primary identified molecular target for type 1 Gaucher disease and other glycosphingolipidoses, appears likely to fulfill high expectations for clinical efficacy...
The cellular pathology of lysosomal diseasesTimothy M Cox
Department of Medicine, University of Cambridge, Cambridge, UK
J Pathol 226:241-54. 2012....
Null alleles of the aldolase B gene in patients with hereditary fructose intoleranceM Ali
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, UK
J Med Genet 31:499-503. 1994....
DNA diagnosis of fatal fructose intolerance from archival tissueM Ali
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, U K
Q J Med 86:25-30. 1993..Analysis of aldolase B genes in this sample by procedures based on the polymerase chain reaction (PCR) confirmed the presence of both mutations in the proposita, the diagnosis of hereditary fructose intolerance, and the cause of death...
Neonatal screening for hereditary fructose intolerance: frequency of the most common mutant aldolase B allele (A149P) in the British populationC L James
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, UK
J Med Genet 33:837-41. 1996..Our findings have implications for establishing an interventional mass screening programme to identify newborn infants with HFI in the UK...
Hereditary fructose intoleranceM Ali
University of Cambridge, Department of Medicine, Addenbrooke s Hospital, UK
J Med Genet 35:353-65. 1998..Here we review the biochemical, genetic, and molecular basis of human aldolase B deficiency in HFI, a disorder which responds to dietary therapy and in which the principal manifestations of disease are thus preventable...
Molecular characterization of a ferrochelatase gene defect causing anomalous RNA splicing in erythropoietic protoporphyriaR P Sarkany
Department of Medicine, University of Cambridge, Addenbrooke s Hospital
J Invest Dermatol 102:481-4. 1994..This leads to the expression of a ferrochelatase protein lacking a central region of 40 amino acids...
Lysosomal delivery of therapeutic enzymes in cell models of Fabry diseaseD Marchesan
Department of Medicine Addenbrooke s Hospital, University of Cambridge, Hills Road, Cambridge, CB2 0QQ, UK
J Inherit Metab Dis 35:1107-17. 2012..If these observations are confirmed in vivo, alternative mechanisms will be needed to explain the ready clearance of storage from endothelial cells in patients undergoing enzyme replacement therapy...
Alteration of substrate specificity by a naturally-occurring aldolase B mutation (Ala337-->Val) in fructose intoleranceP Rellos
Department of Medicine, University of Cambridge, Level 5, Addenbrooke s Hospital, Cambridge CB2 2QQ, UK
Biochem J 340:321-7. 1999....
Clinical evaluation of biomarkers in Gaucher diseaseP B Deegan
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Cambridge, UK
Acta Paediatr Suppl 94:47-50; discussion 37-8. 2005....
Biomarkers in lysosomal storage diseases: a reviewT M Cox
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Cambridge, UK
Acta Paediatr Suppl 94:39-42; discussion 37-8. 2005..Conclusion: New methods for the identification of novel biomarkers have the potential to provide mechanistic insights into the molecular pathogenesis of LSDs, including Fabry disease and Gaucher disease...
The role of the iminosugar N-butyldeoxynojirimycin (miglustat) in the management of type I (non-neuronopathic) Gaucher disease: a position statementT M Cox
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Cambridge, UK
J Inherit Metab Dis 26:513-26. 2003..This position statement represents the consensus viewpoint of an independent international advisory council to the European Working Group on Gaucher Disease...
Acute intermittent porphyria: fatal complications of treatmentP E Stein
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Cambridge
Clin Med 12:293-4. 2012..Intravenous glucose in water solutions are contraindicated as they aggravate hyponatraemia, which can prove fatal...
Tartrate-resistant acid phosphatase (Acp 5): identification in diverse human tissues and dendritic cellsA R Hayman
Department of Medicine, University of Cambridge, Addenbrooke's Hospital, Cambridge, United Kingdom
J Histochem Cytochem 49:675-84. 2001..Our findings demonstrate widespread expression of TRAP in human tissues. Its abundant expression in epithelia and dendritic cells suggests a potential role in antigen processing and in immune responses...
Gaucher disease: understanding the molecular pathogenesis of sphingolipidosesT M Cox
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, UK
J Inherit Metab Dis 24:106-21; discussion 87-8. 2001....
A novel HEXB mutation and its structural effects in juvenile Sandhoff diseaseS Z Wang
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Hills Road, Cambridge CB2 2QQ, UK
Mol Genet Metab 95:236-8. 2008..Identification of D459A contributes to diagnosis and molecular understanding of attenuated Sandhoff disease variants...
Twin pairs showing discordance of phenotype in adult Gaucher's diseaseR H Lachmann
Department of Medicine, University of Cambridge, Addenbrooke's Hospital, Cambridge, UK
QJM 97:199-204. 2004..Understanding the interactions between heritable and non-heritable factors will be critical for an analysis of pathogenesis, and the treatment of individuals predisposed to Gaucher disease...
Autosomal recessive erythropoietic protoporphyria: a syndrome of severe photosensitivity and liver failureR P Sarkany
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, UK
QJM 88:541-9. 1995..Studies of disease inheritance in families affected by protoporphyria may help identify those predisposed to develop severe liver complications, a distinction not currently possible...
Monitoring enzyme replacement therapy in Fabry disease--role of urine globotriaosylceramideP D Whitfield
Biochemical Genetics Unit, Addenbrooke's NHS Trust, Cambridge, UK
J Inherit Metab Dis 28:21-33. 2005....
Aldolase B and fructose intoleranceT M Cox
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, U K
FASEB J 8:62-71. 1994..The incidence of dental caries is consequently much reduced...
Substrate reduction therapy for lysosomal storage diseasesT M Cox
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Cambridge, UK
Acta Paediatr Suppl 94:69-75; discussion 57. 2005..The results of ongoing clinical trials of miglustat in type 3 Gaucher disease, Niemann-Pick disease type C and GM2 gangliosidosis are eagerly awaited...
Quantifying the Erlenmeyer flask deformityA Carter
Department of Radiology, Addenbrooke s Hospital, University of Cambridge, Cambridge, UK
Br J Radiol 85:905-9. 2012..To devise an easily applied definition of this deformity, we investigated a cohort of knee radiographs in which there was consensus between three experienced radiologists as to the presence or absence of Erlenmeyer flask morphology...
Molecular analysis of functional and nonfunctional genes for human ferrochelatase: isolation and characterization of a FECH pseudogene and its sublocalization on chromosome 3D M Whitcombe
Department of Medicine, University of Cambridge, United Kingdom
Genomics 20:482-6. 1994..The existence of the ferrochelatase pseudogene has practical implications for the molecular analysis of mutations responsible for erythropoietic protoporphyria in man...
Classification and genetic features of neonatal haemochromatosis: a study of 27 affected pedigrees and molecular analysis of genes implicated in iron metabolismA L Kelly
Department of Medicine, University of Cambridge, Level 5, Box 157, Addenbrooke's Hospital, Cambridge CB2 2QQ, UK
J Med Genet 38:599-610. 2001....
Widespread expression of tartrate-resistant acid phosphatase (Acp 5) in the mouse embryoA R Hayman
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, UK
J Anat 196:433-41. 2000....
Mice lacking tartrate-resistant acid phosphatase (Acp 5) have disrupted endochondral ossification and mild osteopetrosisA R Hayman
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, UK
Development 122:3151-62. 1996....
Mice lacking tartrate-resistant acid phosphatase (Acp 5) have disordered macrophage inflammatory responses and reduced clearance of the pathogen, Staphylococcus aureusA J Bune
Department of Medicine, University of Cambridge, Addenbrooke s Hospital, Cambridge, UK
Immunology 102:103-13. 2001..Our study shows that TRAP participates in the inflammatory response of the Mphi and influences effector signalling pathways in innate immunity...
Overlapping functions of lysosomal acid phosphatase (LAP) and tartrate-resistant acid phosphatase (Acp5) revealed by doubly deficient miceA Suter
Zentrum Biochemie und Molekulare Zellbiologie, Abt Biochemie II, Universitat Gottingen, Heinrich Düker Weg 12, 37073 Gottingen, Germany
Development 128:4899-910. 2001..We conclude that for several substrates LAP and Acp5 can substitute for each other and that these acid phosphatases are essential for processing of non-collagenous proteins, including osteopontin, by osteoclasts...
Inhibition of substrate synthesis as a strategy for glycolipid lysosomal storage disease therapyF M Platt
Glycobiology Institute, Department of Biochemistry, University of Oxford, UK
J Inherit Metab Dis 24:275-90. 2001..Further trials in type I Gaucher disease are in progress; studies are planned in patients with GSL storage in the CNS...
Sustained therapeutic effects of oral miglustat (Zavesca, N-butyldeoxynojirimycin, OGT 918) in type I Gaucher diseaseD Elstein
Shaare Zedek Medical Centre, Jerusalem, Israel
J Inherit Metab Dis 27:757-66. 2004..In conclusion, miglustat was increasingly effective over time and showed acceptable tolerability in patients who continued with treatment for 3 years...
Type 5 acid phosphatase. Sequence, expression and chromosomal localization of a differentiation-associated protein of the human macrophageD K Lord
Department of Haematology, Royal Postgraduate Medical School, London, England
Eur J Biochem 189:287-93. 1990..Type 5 acid phosphatase thus represents a tightly regulated system for the study of molecular events in the differentiation programme of the normal macrophage...
Calvarial osteoclasts express a higher level of tartrate-resistant acid phosphatase than long bone osteoclasts and activation does not depend on cathepsin K or L activityS Perez-Amodio
Experimental Periodontology, Academic Center for Dentistry Amsterdam, Universiteit van Amsterdam and Vrije Universiteit, Louwesweg 1, 1066 EA Amsterdam, The Netherlands
Calcif Tissue Int 79:245-54. 2006....
