Research Topics
Species | C J SumnerSummaryAffiliation: National Institutes of Health Country: USA Publications
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Detail Information
Publications
A novel cell immunoassay to measure survival of motor neurons protein in blood cellsStephen J Kolb
Howard Hughes Medical Institute and Department of Biochemistry and Biophysics, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania 19104 6148, USA
BMC Neurol 6:6. 2006..A major goal of current therapeutic approaches is to increase SMN levels in SMA patients. The purpose of this study was to develop a reliable assay to measure SMN protein levels from peripheral blood samples...
Jaw drop in Kennedy's diseaseCharlotte J Sumner
Neurogenetics Branch, National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD 20892, USA
Neurology 59:1471-2. 2002
Valproic acid increases SMN levels in spinal muscular atrophy patient cellsCharlotte J Sumner
Neurogenetics Branch, National Institute of Neurologic Diseases and Stroke NIH, Building 10, Room 3B 14, MSC 1250, 10 Center Drive, Bethesda, MD 20892, USA
Ann Neurol 54:647-54. 2003..Valproic acid may increase SMN levels both by activating the SMN promoter and by preventing exon 7 skipping in SMN transcripts. Valproic acid and related compounds warrant further investigation as potential treatment for SMA...
SMN mRNA and protein levels in peripheral blood: biomarkers for SMA clinical trialsC J Sumner
Neurogenetics Branch, National Institute of Neurological Disorders and Stroke, Bethesda, MD 20892, USA
Neurology 66:1067-73. 2006..Clinical trials of drugs that increase SMN protein levels in vitro are currently under way in patients with spinal muscular atrophy...
Targeting splicing in spinal muscular atrophyBarrington G Burnett
Ann Neurol 63:3-6. 2008
Molecular mechanisms of spinal muscular atrophyCharlotte J Sumner
Department of Neurology, Johns Hopkins University, Baltimore, Maryland 21287, USA
J Child Neurol 22:979-89. 2007..Histone deacetylase inhibitors will be discussed as an example...
Safety, tolerability, and pharmacokinetics of high-dose idebenone in patients with Friedreich ataxiaNicholas A Di Prospero
Neurogenetics Branch, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, MD 20892 3705, USA
Arch Neurol 64:803-8. 2007..Some studies suggest that higher doses of idebenone may be more effective, but pharmacology and toxicology at higher doses have not been investigated in human beings...
Trichostatin A increases SMN expression and survival in a mouse model of spinal muscular atrophyAmy M Avila
Neurogenetics Branch, National Institute of Neurological Disorders and Stroke NINDS, NIH, Bethesda, MA 20892, USA
J Clin Invest 117:659-71. 2007..These results indicate that the hydroxamic acid class of HDAC inhibitors activates SMN2 gene expression in vivo and has an ameliorating effect on the SMA disease phenotype when administered after disease onset...
Therapeutics development for spinal muscular atrophyCharlotte J Sumner
Neurogenetics Branch, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland 20892, USA
NeuroRx 3:235-45. 2006..Early clinical trials of candidate therapeutics are now ongoing in SMA patients. Clinical trials in this disease present a unique set of challenges, including the development of meaningful outcome measures and disease biomarkers...
The role of histone acetylation in SMN gene expressionLauren E Kernochan
Neurogenetics Branch, National Institute of Neurological Disorders and Stroke, Bethesda, MD 20892, USA
Hum Mol Genet 14:1171-82. 2005..These data indicate that histone acetylation modulates SMN gene expression and that pharmacological manipulation of this epigenetic determinant is feasible. HDAC2, in particular, may be a future therapeutic target for SMA...
Indoprofen upregulates the survival motor neuron protein through a cyclooxygenase-independent mechanismMitchell R Lunn
Department of Biological Sciences, Columbia University, Fairchild Center, MC 2406, 1212 Amsterdam Avenue, New York, NY 10027, USA
Chem Biol 11:1489-93. 2004..No other NSAIDs or COX inhibitors tested exhibited this activity...
Lack of spartin protein in Troyer syndrome: a loss-of-function disease mechanism?Joanna C Bakowska
Cellular Neurology Unit, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland 20892 3704, USA
Arch Neurol 65:520-4. 2008..This mutation is predicted to cause premature termination of the spartin protein. However, it remains unknown whether this truncated spartin protein is absent or is present and partially functional in patients...
