Literature DB >> 4522795

Globoid cell leukodystrophy: deficiency of lactosyl ceramide beta-galactosidase.

D A Wenger, M Sattler, W Hiatt.   

Abstract

Activity of lactosyl ceramide beta-galactosidase (beta-D-galactoside galactohydrolase, EC 3.2.1.23) was found to be extremely low in enzyme preparations from liver, brain, and cultured skin fibroblasts from patients with Krabbe's disease. Leukocytes from one set of parents had enzyme levels approximately half those measured in control leukocytes. The low activity observed for this galactolipid hydrolase is the fourth enzymatic deficiency noted for this genetic disease. Beta-galactosidase activity toward galactocerebroside, psychosine, and monogalactosyl diglyceride is also low in patients with Krabbe's disease. Other lysosomal enzymes measured were found to be in the normal range. This enzymatic defect may provide a better explanation for the pathological and chemical findings previously reported for this syndrome.

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Year:  1974        PMID: 4522795      PMCID: PMC388113          DOI: 10.1073/pnas.71.3.854

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  21 in total

1.  Globoid cell leukodystrophy (Krabbe's disease): morphological and biochemical studies.

Authors:  J M Andrews; P A Cancilla; J Grippo; J H Menkes
Journal:  Neurology       Date:  1971-04       Impact factor: 9.910

2.  Deficiency of monogalactosyl diglycerid beta-B-galactosidase activity in krabbe's disease.

Authors:  D A Wenger; M Satter; J P Markey
Journal:  Biochem Biophys Res Commun       Date:  1973-07-17       Impact factor: 3.575

3.  Galactosylsphingosine galactosyl hydrolase. Partial purification and properties of the enzyme in rat brain.

Authors:  T Miyatake; K Suzuki
Journal:  J Biol Chem       Date:  1972-09-10       Impact factor: 5.157

4.  Glycolipids of the brain in gargoylism.

Authors:  T Taketomi; T Yamakawa
Journal:  Jpn J Exp Med       Date:  1967-02

5.  Studies in globoid (Krabbe) leukodystrophy (GLD). V. Controlled enzymic studies in ten human cases.

Authors:  J Austin; K Suzuki; D Armstrong; R Brady; B K Bachhawat; J Schlenker; D Stumpf
Journal:  Arch Neurol       Date:  1970-12

6.  Krabbe's globoid cell leukodystrophy: deficiency of glactocerebrosidase in serum, leukocytes, and fibroblasts.

Authors:  Y Suzuki; K Suzuki
Journal:  Science       Date:  1971-01-08       Impact factor: 47.728

7.  Lactosylceramidosis: lactosylceramide galactosyl hydrolase deficiency and accumulation of lactosylceramide in cultured skin fibroblasts.

Authors:  G Dawson; R Matalon; A O Stein
Journal:  J Pediatr       Date:  1971-09       Impact factor: 4.406

8.  Lactosyl ceramidosis: catabolic enzyme defect of glycosphingolipid metabolism.

Authors:  G Dawson; A O Stein
Journal:  Science       Date:  1970-10-30       Impact factor: 47.728

9.  Globoid cell leucodystrophy (Krabbe's disease): deficiency of galactocerebroside beta-galactosidase.

Authors:  K Suzuki; Y Suzuki
Journal:  Proc Natl Acad Sci U S A       Date:  1970-06       Impact factor: 11.205

10.  Globoid cell leukodystrophy: additional deficiency of psychosine galactosidase.

Authors:  T Miyatake; K Suzuki
Journal:  Biochem Biophys Res Commun       Date:  1972-08-07       Impact factor: 3.575

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  24 in total

Review 1.  Biochemistry and genetics of gangliosidoses.

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

2.  Prevalence of different types of lysosomal storage diseases in Saudi Arabia.

Authors:  P T Ozand; G Gascon; A al Aqeel; G Roberts; M Dhalla; S B Subramanyam
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

3.  Krabbe's disease. A light and electron microscopic study.

Authors:  S S Schochet; W F McCormick; G F Powell
Journal:  Acta Neuropathol       Date:  1976-10-15       Impact factor: 17.088

Review 4.  Glycosphingolipid hydrolases: properties and molecular genetics.

Authors:  M Wan Ho; A G Norden; J A Alhadeff; J S O'Brien
Journal:  Mol Cell Biochem       Date:  1977-10-07       Impact factor: 3.396

Review 5.  Progress in investigations of sphingolipidoses.

Authors:  M Adachi; L Schneck; B W Volk
Journal:  Acta Neuropathol       Date:  1978-08-07       Impact factor: 17.088

6.  Measurement of psychosine in dried blood spots--a possible improvement to newborn screening programs for Krabbe disease.

Authors:  Coleman T Turgeon; Joseph J Orsini; Karen A Sanders; Mark J Magera; Thomas J Langan; Maria L Escolar; Patricia Duffner; Devin Oglesbee; Dimitar Gavrilov; Silvia Tortorelli; Piero Rinaldo; Kimiyo Raymond; Dietrich Matern
Journal:  J Inherit Metab Dis       Date:  2015-03-12       Impact factor: 4.982

Review 7.  Large animal models of neurological disorders for gene therapy.

Authors:  Christine Gagliardi; Bruce A Bunnell
Journal:  ILAR J       Date:  2009

8.  The interrelations between high- and low-molecular weight forms of normal and mutant (Krabbe-disease) galactocerebrosidase.

Authors:  Y Ben-Yoseph; M Hungerford; H L Nadler
Journal:  Biochem J       Date:  1980-07-01       Impact factor: 3.857

9.  Krabbe's globoid cell leucodystrophy. Studies on galactosylceramide beta-galactosidase and non-specific beta-galactosidase of leucocytes, cultured skin fibroblasts, and amniotic fluid cells.

Authors:  G T Besley; A D Bain
Journal:  J Med Genet       Date:  1976-06       Impact factor: 6.318

10.  The twitcher mouse. A model of human globoid cell leukodystrophy (krabbe's disease).

Authors:  K Suzuki; K Suzuki
Journal:  Am J Pathol       Date:  1983-06       Impact factor: 4.307

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