Literature DB >> 7327559

beta-Galactosidase-neuraminidase deficiency in adults: deficiency of a freeze-labile neuraminidase in leukocytes and fibroblasts.

Y Suzuki, H Sakuraba, M Potier, M Akagi, M Sakai, H Beppu.   

Abstract

4-methylumbelliferyl neuraminidase activity was studied in fibroblasts, leukocytes, and frozen tissues from adult patients with beta-galactosidase-neuraminidase deficiency and specific clinical manifestations. This enzyme was almost completely deficient in fibroblasts, but the residual activity was relatively high (20% of the control mean) in the leukocytes from the patients. The frozen liver from one patient showed the enzyme activity as high as controls. This enzyme consisted of two components, freeze-labile and freeze-stable, and it was demonstrated that only the labile enzyme was deficient in fibroblasts and leukocytes. The apparently normal activity of neuraminidase in frozen autopsy tissues of a patient may be explained by the loss of the labile component in control tissues after a long-term freezing. The neuraminidase activity was variable in parents and no definite conclusion was drawn on the hereditary nature of the disease.

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Year:  1981        PMID: 7327559     DOI: 10.1007/bf00282820

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  15 in total

1.  Macular cherry-red spots and myoclonus with dementia: coexistent neuraminidase and beta-galactosidase deficiencies.

Authors:  D A Wenger; T J Tarby; C Wharton
Journal:  Biochem Biophys Res Commun       Date:  1978-05-30       Impact factor: 3.575

2.  Fluorometric assay of neuraminidase with a sodium (4-methylumbelliferyl-alpha-D-N-acetylneuraminate) substrate.

Authors:  M Potier; L Mameli; M Bélisle; L Dallaire; S B Melançon
Journal:  Anal Biochem       Date:  1979-04-15       Impact factor: 3.365

Review 3.  The use of white cells as a source of diagnostic material for lipid storage diseases.

Authors:  R A Snyder; R O Brady
Journal:  Clin Chim Acta       Date:  1969-08       Impact factor: 3.786

4.  Macular cherry-red spot, corneal clouding, and beta-galactosidase deficiency. Clinical, biochemical, and electron microscopic study of a new autosomal recessive storage disease.

Authors:  M F Goldberg; E Cotlier; L G Fichenscher; K Kenyon; R Enat; S A Borowsky
Journal:  Arch Intern Med       Date:  1971-09

5.  Adult type mucolipidosis with beta-galactosidase and sialidase deficiency. Histological and biochemical studies.

Authors:  T Kobayashi; M Ohta; I Goto; Y Tanaka; Y Kuroiwa
Journal:  J Neurol       Date:  1979-09       Impact factor: 4.849

6.  Neuraminidase activity in the mucolipidoses (types I, II and III) and the cherry-red spot myoclonus syndrome.

Authors:  M Potier; G Beauregard; M Bélisle; L Mameli; V N Hong; S B Melançon; L Dallaire
Journal:  Clin Chim Acta       Date:  1979-12-03       Impact factor: 3.786

7.  beta-Galactosidase deficiency in juvenile and adult patients. Report of six Japanese cases and review of literature.

Authors:  Y Suzuki; N Nakamura; K Fukuoka; Y Shimada; M Uono
Journal:  Hum Genet       Date:  1977-04-15       Impact factor: 4.132

8.  Macular cherry-red spots and beta-galactosidase deficiency in an adult. An autopsy case with progressive cerebellar ataxia, myoclonus, thrombocytopathy, and accumulation of polysaccharide in liver.

Authors:  Y Suzuki; N Nakamura; Y Shimada; H Yotsumoto; H Endo; K Nagashima
Journal:  Arch Neurol       Date:  1977-03

9.  Studies on the sialidoses: properties of human leucocyte neuraminidases.

Authors:  V N Hong; G Beauregard; M Potier; M Bélisle; L Mameli; R Gatti; P Durand
Journal:  Biochim Biophys Acta       Date:  1980-12-04

10.  Neuraminidase deficiency in the original patient with the Goldberg syndrome.

Authors:  G H Thomas; M F Goldberg; C S Miller; L W Reynolds
Journal:  Clin Genet       Date:  1979-11       Impact factor: 4.438

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  3 in total

1.  Sialidosis and galactosialidosis: chromosomal assignment of two genes associated with neuraminidase-deficiency disorders.

Authors:  O T Mueller; W M Henry; L L Haley; M G Byers; R L Eddy; T B Shows
Journal:  Proc Natl Acad Sci U S A       Date:  1986-03       Impact factor: 11.205

2.  Biochemical study of sialidosis type I in a Russian family.

Authors:  I V Tsvetkova; N A Petushkova; T V Zolotuchina; V I Kucharenko; E L Rosenfeld
Journal:  J Inherit Metab Dis       Date:  1987       Impact factor: 4.982

3.  Freeze-stable sialidase activity in human leucocytes: substrate specificity, inhibitor susceptibility, detergent requirements and subcellular localization.

Authors:  P J Waters; A P Corfield; R Eisenthal; C A Pennock
Journal:  Biochem J       Date:  1994-08-01       Impact factor: 3.857

  3 in total

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