Literature DB >> 16079982

Proton MR spectroscopy in three children with Tay-Sachs disease.

Kubilay Aydin1, Baris Bakir, Burak Tatli, Ege Terzibasioglu, Meral Ozmen.   

Abstract

BACKGROUND: Tay-Sachs disease is an inherited metabolic disease caused by the accumulation of GM(2) gangliosides in the central nervous system. Deficiency of hexosaminidase A leads to the accumulation of gangliosides in neurons, axons and glial cells.
OBJECTIVE: To present the cranial MRI and proton MR spectroscopy findings of children of Tay-Sachs disease.
MATERIALS AND METHODS: Three children aged 10, 20 and 21 months were examined.
RESULTS: On T2-weighted MR images there were hyperintense signal changes in the basal ganglia and cerebral white matter. MR spectroscopy demonstrated an increase in myoinositol/creatine and choline/creatine ratios with a decrease in the N-acetyl aspartate/creatine ratio.
CONCLUSIONS: The spectroscopy findings support demyelination, gliosis and neuronal loss in the neuropathological process of Tay-Sachs disease.

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Year:  2005        PMID: 16079982     DOI: 10.1007/s00247-005-1542-3

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  9 in total

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Journal:  Neuroradiology       Date:  1992       Impact factor: 2.804

2.  Histological observation of the brain of Tay-Sachs disease with seizure and chronic DPH intoxication--report of an autopsy case.

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Journal:  AJNR Am J Neuroradiol       Date:  1990 Jan-Feb       Impact factor: 3.825

5.  Inflammatory CNS demyelination: histopathologic correlation with in vivo quantitative proton MR spectroscopy.

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Journal:  AJNR Am J Neuroradiol       Date:  1999-10       Impact factor: 3.825

6.  Fine needle aspiration cytology of Tay-Sachs disease. A case report.

Authors:  A Ul Haque
Journal:  Acta Cytol       Date:  1995 Jul-Aug       Impact factor: 2.319

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Authors:  A Brand; C Richter-Landsberg; D Leibfritz
Journal:  Dev Neurosci       Date:  1993       Impact factor: 2.984

8.  Sphingolipidoses in Turkey.

Authors:  Hatice Asuman Ozkara; Meral Topçu
Journal:  Brain Dev       Date:  2004-09       Impact factor: 1.961

Review 9.  A review of chemical issues in 1H NMR spectroscopy: N-acetyl-L-aspartate, creatine and choline.

Authors:  B L Miller
Journal:  NMR Biomed       Date:  1991-04       Impact factor: 4.044

  9 in total
  9 in total

Review 1.  The natural history of juvenile or subacute GM2 gangliosidosis: 21 new cases and literature review of 134 previously reported.

Authors:  Gustavo H B Maegawa; Tracy Stockley; Michael Tropak; Brenda Banwell; Susan Blaser; Fernando Kok; Roberto Giugliani; Don Mahuran; Joe T R Clarke
Journal:  Pediatrics       Date:  2006-10-02       Impact factor: 7.124

2.  Serial 1H-MRS in GM2 gangliosidoses.

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Journal:  Eur J Pediatr       Date:  2007-03-27       Impact factor: 3.183

Review 3.  Decreased T2 signal in the thalami may be a sign of lysosomal storage disease.

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4.  Natural history of Tay-Sachs disease in sheep.

Authors:  Brett Story; Toloo Taghian; Jillian Gallagher; Jey Koehler; Amanda Taylor; Ashley Randle; Kayly Nielsen; Amanda Gross; Annie Maguire; Sara Carl; Siauna Johnson; Deborah Fernau; Elise Diffie; Paul Cuddon; Carly Corado; Sundeep Chandra; Miguel Sena-Esteves; Edwin Kolodny; Xuntian Jiang; Douglas Martin; Heather Gray-Edwards
Journal:  Mol Genet Metab       Date:  2021-08-21       Impact factor: 4.204

Review 5.  In Vivo NMR Studies of the Brain with Hereditary or Acquired Metabolic Disorders.

Authors:  Erica B Sherry; Phil Lee; In-Young Choi
Journal:  Neurochem Res       Date:  2015-11-26       Impact factor: 3.996

6.  Detection and characterization of neurotoxicity in cancer patients using proton MR spectroscopy.

Authors:  Emilie A Steffen-Smith; Pamela L Wolters; Paul S Albert; Eva H Baker; Kim C Shimoda; Alan S Barnett; Katherine E Warren
Journal:  Childs Nerv Syst       Date:  2008-02-22       Impact factor: 1.475

Review 7.  Genetics and Therapies for GM2 Gangliosidosis.

Authors:  Maria Begona Cachon-Gonzalez; Eva Zaccariotto; Timothy Martin Cox
Journal:  Curr Gene Ther       Date:  2018       Impact factor: 4.391

8.  Magnetic resonance imaging and spectroscopy in late-onset GM2-gangliosidosis.

Authors:  Olivia E Rowe; D Rangaprakash; Akila Weerasekera; Neha Godbole; Elizabeth Haxton; Peter F James; Christopher D Stephen; Robert L Barry; Florian S Eichler; Eva-Maria Ratai
Journal:  Mol Genet Metab       Date:  2021-06-24       Impact factor: 4.204

9.  7T MRI Predicts Amelioration of Neurodegeneration in the Brain after AAV Gene Therapy.

Authors:  Heather L Gray-Edwards; Anne S Maguire; Nouha Salibi; Lauren E Ellis; Taylor L Voss; Elise B Diffie; Jey Koehler; Ashley N Randle; Amanda R Taylor; Brandon L Brunson; Thomas S Denney; Ronald J Beyers; Atoska S Gentry; Amanda L Gross; Ana R Batista; Miguel Sena-Esteves; Douglas R Martin
Journal:  Mol Ther Methods Clin Dev       Date:  2019-12-24       Impact factor: 6.698

  9 in total

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