Literature DB >> 6446239

Morquio syndrome (mucopolysaccharidosis IV B) associated with beta-galactosidase deficiency. Report of two cases.

H Groebe, M Krins, H Schmidberger, K von Figura, K Harzer, H Kresse, E Paschke, A Sewell, K Ullrich.   

Abstract

Two male patients, aged 6 and 25, both with normal intelligence and absence of neurological abnormalities, exhibited dysostosis multiplex, dwarfism, odontoid anomalies, cloudy corneas, exessive excretion of keratan sulfate, and abnormal urinary oligosaccharides. Leukocytes and fibroblasts of both patients were deficient in acid beta-galactosidase (beta-gal) and normal in N-acetylgalactosamine-6-sulfate sulfatase, the deficient enzyme in classical Morquio syndrome. The beta-gal deficiency was not due to an endogenous inhibitor, and the parents exhibited intermediate activities. Deficient beta-gal activity was observed toward p-nitrophenyl-beta-galactoside, 4-methylumbelliferyl-beta-galactoside (4 MU-beta-gal), lactose, GM1 ganglioside, keratan sulfate, and asialofetuin (ASF). Under standard assay conditions, the residual activity was similar for all substrates tested. Toward p-nitrophenyl-beta-glactoside, the mutant enzyme behaved as a Km variant.

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Year:  1980        PMID: 6446239      PMCID: PMC1686002     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  24 in total

1.  The existence of a second route for the transfer of certain glycoproteins from the circulation into the liver.

Authors:  R J Stockert; A G Morell; I H Scheinberg
Journal:  Biochem Biophys Res Commun       Date:  1976-02-09       Impact factor: 3.575

2.  Enzymatic methods for the determination of small quantities of isomeric chondroitin sulfates.

Authors:  H Saito; T Yamagata; S Suzuki
Journal:  J Biol Chem       Date:  1968-04-10       Impact factor: 5.157

3.  Sequential thin layer chromatography of urinary acidic glycosaminglycans.

Authors:  R Humbel; N A Chamoles
Journal:  Clin Chim Acta       Date:  1972-08       Impact factor: 3.786

4.  -galactosidase activity of human skin fibroblasts.

Authors:  J N Kanfer; C Spielvogel
Journal:  Biochim Biophys Acta       Date:  1972-12-07

5.  Differential effect of chloride ions on -galactosidase isoenzymes: a method for separate assay.

Authors:  M W Ho; J S O'Brien
Journal:  Clin Chim Acta       Date:  1971-05       Impact factor: 3.786

6.  Morquio's syndrome: deficiency of a chondroitin sulfate N-acetylhexosamine sulfate sulfatase.

Authors:  R Matalon; B Arbogast; P Justice; I K Brandt; A Dorfman
Journal:  Biochem Biophys Res Commun       Date:  1974-11-27       Impact factor: 3.575

7.  The electrophoretic separation of human -galactosidases on cellulose acetate.

Authors:  A L Fluharty; E L Lassila; M T Porter; H Kihara
Journal:  Biochem Med       Date:  1971-04

8.  Physical and chemical studies on ceruloplasmin. IV. Preparation of radioactive, sialic acid-free ceruloplasmin labeled with tritium on terminal D-galactose residues.

Authors:  A G Morell; C J Van den Hamer; I H Scheinberg; G Ashwell
Journal:  J Biol Chem       Date:  1966-08-25       Impact factor: 5.157

9.  Spondyloepiphyseal dysplasia, corneal clouding, normal intelligence and acid beta-galactosidase deficiency.

Authors:  J S O'Brien; E Gugler; A Giedion; U Wiessmann; N Herschkowitz; C Meier; J Leroy
Journal:  Clin Genet       Date:  1976-05       Impact factor: 4.438

10.  Generalized gangliosidosis: beta-galactosidase deficiency.

Authors:  S Okada; J S O'Brien
Journal:  Science       Date:  1968-05-31       Impact factor: 47.728

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  22 in total

1.  Morquio-B syndrome (MPS-IV B) associated with beta-galactosidase deficiency in two siblings.

Authors:  Jayesh J Sheth; Frenny J Sheth; Raktima Bhattacharya
Journal:  Indian J Pediatr       Date:  2002-01       Impact factor: 1.967

2.  Anesthetic management for foramen magnum decompression in a patient with Morquio syndrome: a case report.

Authors:  Lakshminarsimhaiah Geetha; Muthuchellappan Radhakrishnan; Biligiri Sridhar Raghavendra; G S Umamaheswara Rao; Bhagavatula Indira Devi
Journal:  J Anesth       Date:  2010-06-03       Impact factor: 2.078

3.  Ganglioside GM1 metabolism in living human fibroblasts with beta-galactosidase deficiency.

Authors:  G M Mancini; A T Hoogeveen; H Galjaard; J E Mansson; L Svennerholm
Journal:  Hum Genet       Date:  1986-05       Impact factor: 4.132

4.  Ocular histopathology and ultrastructure of Morquio syndrome (systemic mucopolysaccharidosis IV A).

Authors:  M Iwamoto; Y Nawa; I H Maumenee; J Young-Ramsaran; R Matalon; W R Green
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  1990       Impact factor: 3.117

5.  Fluorous iminoalditols act as effective pharmacological chaperones against gene products from GLB₁ alleles causing GM1-gangliosidosis and Morquio B disease.

Authors:  Katrin M Fantur; Tanja M Wrodnigg; Arnold E Stütz; Bettina M Pabst; Eduard Paschke
Journal:  J Inherit Metab Dis       Date:  2011-10-28       Impact factor: 4.982

Review 6.  Screening for lysosomal disorders.

Authors:  K Ullrich
Journal:  Eur J Pediatr       Date:  1994       Impact factor: 3.183

Review 7.  Ganglioside storage diseases: an updated review.

Authors:  J S O'Brien
Journal:  Ital J Neurol Sci       Date:  1981-08

8.  The mucopolysaccharidoses: biochemistry and clinical symptoms.

Authors:  H Kresse; M Cantz; K von Figura; J Glössl; E Paschke
Journal:  Klin Wochenschr       Date:  1981-08-17

9.  The simple detection of neuraminic acid-containing urinary oligosaccharides in patients with glycoprotein storage diseases.

Authors:  A C Sewell
Journal:  J Inherit Metab Dis       Date:  1983       Impact factor: 4.982

10.  Morquio's disease type A: absence of material cross reacting with antibodies against N-acetylgalactosamine-6-sulfate sulfatase.

Authors:  J Glössl; K Lembeck; G Gamse; H Kresse
Journal:  Hum Genet       Date:  1980       Impact factor: 4.132

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