Enhanced survival in Sandhoff disease mice receiving a combination of substrate deprivation therapy and bone marrow transplantation (original) (raw)

Delayed symptom onset and increased life expectancy in Sandhoff disease mice treated with N-butyldeoxynojirimycin

Raymond Dwek

Proceedings of the National Academy of Sciences, 1999

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Improved outcome of N-butyldeoxygalactonojirimycin-mediated substrate reduction therapy in a mouse model of Sandhoff disease

Raymond Dwek

Neurobiology of Disease, 2004

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Introduction of an N-Glycan Sequon Into HEXA Enhances Human β-Hexosaminidase Cellular Uptake in a Model of Sandhoff Disease

Fumiko Matsuzawa

Molecular Therapy, 2010

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Substrate deprivation therapy in juvenile Sandhoff disease

Michel Willemsen

Journal of Inherited Metabolic Disease, 2009

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Establishment of immortalized Schwann cells from Sandhoff mice and corrective effect of recombinant human b-hexosaminidase A on the accumulated GM2 ganglioside

Kohji Itoh

J Hum Genet, 2005

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Establishment of immortalized Schwann cells from Sandhoff mice and corrective effect of recombinant human β-hexosaminidase A on the accumulated GM2 ganglioside

Kohji Itoh

Journal of Human Genetics, 2005

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Beneficial Effects of Acetyl-DL-Leucine (ADLL) in a Mouse Model of Sandhoff Disease

Barry Boland

Journal of Clinical Medicine

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A Pro504 right-arrow Ser Substitution in the beta -Subunit of beta -Hexosaminidase A Inhibits alpha -Subunit Hydrolysis of GM2 Ganglioside, Resulting in Chronic Sandhoff Disease

Aleksander Hinek

Journal of Biological Chemistry, 1998

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Conditional expression of human β-hexosaminidase in the neurons of Sandhoff disease rescues mice from neurodegeneration but not neuroinflammation

John Olschowka

Journal of Neuroinflammation, 2012

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Efficacy of a Bicistronic Vector for Correction of Sandhoff Disease in a Mouse Model

Patrick Thompson

Molecular Therapy - Methods & Clinical Development, 2019

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Intravenous administration of scAAV9-Hexb normalizes lifespan and prevents pathology in Sandhoff disease mice

Ganna Panasyuk

Human molecular genetics, 2018

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Long-Term Correction of Sandhoff Disease Following Intravenous Delivery of rAAV9 to Mouse Neonates

Alexander ulloa bello

Molecular Therapy, 2014

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NSAIDs increase survival in the Sandhoff disease mouse: Synergy withN-butyldeoxynojirimycin

Ian Williams

Annals of Neurology, 2004

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Inhibition of substrate synthesis as a strategy for glycolipid lysosomal storage disease therapy

Raymond Dwek

Journal of Inherited Metabolic Disease, 2001

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Dramatically Different Phenotypes in Mouse Models of Human Tay-Sachs and Sandhoff Diseases

Carlos Galán Morales

Human Molecular Genetics, 1996

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126. Comparative Analysis of brain lipids in Mice, Cats, and Humans with Sandhoff disease

Thomas Seyfried

Molecular Genetics and Metabolism, 2009

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Molecular basis of an adult form of Sandhoff disease: Substitution of glutamine for arginine at position 505 of the β-chain of β-hexosaminidase results in a labile enzyme

Vianney de Jong

Biochimica et Biophysica Acta (BBA) - Molecular Basis of Disease, 1993

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Lysosomal Dysfunction in a Mouse Model of Sandhoff Disease Leads to Accumulation of Ganglioside-Bound Amyloid- Peptide

anthony stevens

Journal of Neuroscience, 2012

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Substrate deprivation therapy: a new hope for patients suffering from neuronopathic forms of inherited lysosomal storage diseases

Grzegorz Węgrzyn

Journal of Applied Genetics, 2007

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Innovative Therapeutic Approaches for Improving Patient Life Condition with a Neurological Lysosomal Disease

magali richard

Latest Findings in Intellectual and Developmental Disabilities Research, 2012

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Lyso-GM2 Ganglioside: A Possible Biomarker of Tay-Sachs Disease and Sandhoff Disease

Daisuke Tsuji, Kohji Itoh

PLoS ONE, 2011

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AAV-Mediated Gene Delivery in a Feline Model of Sandhoff Disease Corrects Lysosomal Storage in the Central Nervous System

Judith Hudson, Aime Johnson

ASN neuro, 2015

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Systemic Gene Transfer of a Hexosaminidase Variant Using a scAAV9.47 Vector Corrects GM2 Gangliosidosis in Sandhoff Mice

Patrick Thompson

Human gene therapy, 2016

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Peripheral nervous system manifestations in a Sandhoff disease mouse model: nerve conduction, myelin structure, lipid analysis

Daniel Kirschner

Journal of Negative Results in BioMedicine, 2007

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Occurrence of an anomalous endocytic compartment in fibroblasts from Sandhoff disease patients

Virginia Loredana

Molecular and Cellular Biochemistry, 2010

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Biochemical and pathological evaluation of long-lived mice with globoid cell leukodystrophy after bone marrow transplantation

Marie Vanier

Molecular Genetics and Metabolism, 2005

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