Literature DB >> 404709

GM2 ganglioside lysosomal storage disease in cats with beta-hexosaminidase deficiency.

L C Cork, J F Munnell, M D Lorenz, J V Murphy, H J Baker, M C Rattazzi.   

Abstract

Two kitteens with progressive neurologic disease had increased concentrations of GM2 ganglioside in their cerebral cortex. Examination under the light microscope revealed cytoplasmic vacuolation of neurons and hepatocytes. Transmission and scanning electron microscopy demosntrated cytoplasmic inclusions encompassed by membranes in various central nervous system cell types and in hepatocytes. Beta-D-N-acetyl-hexosaminidase activity was reduced to about 1.0 percent of normal in brain, liver, and cultured skin fibroblasts of the diseased kittens; both major electrophoretic forms, A and B, of the enzyme were deficient. In fibroblasts from the parents of the diseased kittens, this enzyme activity was intermediate between that of affected and normal cats, suggesting an autosomal recessive mode of inheritance of the enzyme defect. Histopahtological and ultrastructural lesions, glycolipid storage, enzyme defect, and pattern of inheritance are similar to those of human GM2 gangliosidosis type 2.

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Year:  1977        PMID: 404709     DOI: 10.1126/science.404709

Source DB:  PubMed          Journal:  Science        ISSN: 0036-8075            Impact factor:   47.728


  41 in total

Review 1.  Biochemistry and genetics of gangliosidoses.

Authors:  K Sandhoff; H Christomanou
Journal:  Hum Genet       Date:  1979       Impact factor: 4.132

2.  Effective gene therapy in an authentic model of Tay-Sachs-related diseases.

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3.  Abnormal epiphyseal development in a feline model of Sandhoff disease.

Authors:  Margaret A McNulty; Patricia B Prevatt; Elizabeth R Nussbaum; Ashley N Randle; Aime K Johnson; Judith A Hudson; Heather L Gray-Edwards; Miguel Sena-Esteves; Douglas R Martin; Cathy S Carlson
Journal:  J Orthop Res       Date:  2020-07-29       Impact factor: 3.494

4.  GM2 gangliosidosis in a Japanese spaniel.

Authors:  J F Cummings; P A Wood; S U Walkley; A de Lahunta; M E DeForest
Journal:  Acta Neuropathol       Date:  1985       Impact factor: 17.088

5.  AAV-mediated gene delivery attenuates neuroinflammation in feline Sandhoff disease.

Authors:  Allison M Bradbury; Tiffany A Peterson; Amanda L Gross; Stephen Z Wells; Victoria J McCurdy; Karen G Wolfe; John C Dennis; Brandon L Brunson; Heather Gray-Edwards; Ashley N Randle; Aime K Johnson; Edward E Morrison; Nancy R Cox; Henry J Baker; Miguel Sena-Esteves; Douglas R Martin
Journal:  Neuroscience       Date:  2016-10-26       Impact factor: 3.590

6.  High resolution MRI anatomy of the cat brain at 3 Tesla.

Authors:  Heather L Gray-Edwards; Nouha Salibi; Eleanor M Josephson; Judith A Hudson; Nancy R Cox; Ashley N Randle; Victoria J McCurdy; Allison M Bradbury; Diane U Wilson; Ronald J Beyers; Thomas S Denney; Douglas R Martin
Journal:  J Neurosci Methods       Date:  2014-02-10       Impact factor: 2.390

7.  Characterization of a new model of GM2-gangliosidosis (Sandhoff's disease) in Korat cats.

Authors:  E A Neuwelt; W G Johnson; N K Blank; M A Pagel; C Maslen-McClure; M J McClure; P M Wu
Journal:  J Clin Invest       Date:  1985-08       Impact factor: 14.808

8.  Neuropathology of mice with targeted disruption of Hexa gene, a model of Tay-Sachs disease.

Authors:  M Taniike; S Yamanaka; R L Proia; C Langaman; T Bone-Turrentine; K Suzuki
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9.  A canine model of human alpha-L-iduronidase deficiency.

Authors:  E Spellacy; R M Shull; G Constantopoulos; E F Neufeld
Journal:  Proc Natl Acad Sci U S A       Date:  1983-10       Impact factor: 11.205

10.  Ectopic dendrites occur only on cortical pyramidal cells containing elevated GM2 ganglioside in alpha-mannosidosis.

Authors:  L A Goodman; P O Livingston; S U Walkley
Journal:  Proc Natl Acad Sci U S A       Date:  1991-12-15       Impact factor: 11.205

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