Literature DB >> 3939537

Diagnosis of classical Morquio's disease: N-acetylgalactosamine 6-sulphate sulphatase activity in cultured fibroblasts, leukocytes, amniotic cells and chorionic villi.

M Yuen, A H Fensom.   

Abstract

The tritiated disulphated trisaccharide 6-sulpho-N-acetylgalactosamine-glucuronic acid-6-sulpho-N-acetyl-[1-3H]galactosaminitol was prepared from chondroitin 6-sulphate for use as a substrate for N-acetylgalactosamine 6-sulphate sulphatase. The reaction product was separated on ECTEOLA cellulose rather than Dowex 1 X 2. The mean activities of normal fibroblasts, leukocytes and amniotic cells were 8.43, 2.59 and 3.14 nmol h-1 mg protein-1, respectively. Fibroblasts from five patients with classical Morquio's disease (mucopolysaccharidosis IVA; MPSIVA) and one patient with neonatal multiple sulphatase deficiency displayed activities of less than 5% of control mean. Activity in amniotic cells from a pregnancy where the fetus was affected with MPS IVA was 6% of control mean. Activity was also found to be present in normal specimens of chorionic villi (mean value 1.21 nmol h-1 mg protein-1), demonstrating the feasibility of first trimester prenatal diagnosis of MPS IVA by assay of activity in this tissue.

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Year:  1985        PMID: 3939537     DOI: 10.1007/bf01801671

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  12 in total

1.  A modified uronic acid carbazole reaction.

Authors:  T BITTER; H M MUIR
Journal:  Anal Biochem       Date:  1962-10       Impact factor: 3.365

2.  Protein measurement with the Folin phenol reagent.

Authors:  O H LOWRY; N J ROSEBROUGH; A L FARR; R J RANDALL
Journal:  J Biol Chem       Date:  1951-11       Impact factor: 5.157

3.  Prenatal diagnosis of mucopolysaccharidoses.

Authors:  P Whiteman
Journal:  Lancet       Date:  1973-06-02       Impact factor: 79.321

4.  The enzymic defect in Morquio's disease: the specificity of N-acetylhexosamine sulfatases.

Authors:  A L Horwitz; A Dorfman
Journal:  Biochem Biophys Res Commun       Date:  1978-02-28       Impact factor: 3.575

5.  First-trimester diagnosis on chorionic villi obtained by direct vision technique.

Authors:  B Gustavii; M A Chester; H Edvall; S Iosif; U Kristoffersson; L Löfberg; A Mineur; F Mitelman
Journal:  Hum Genet       Date:  1984       Impact factor: 4.132

6.  Detection of Morquio A syndrome using radiolabelled substrates derived from keratan sulphate for the estimation of galactose 6-sulphate sulphatase.

Authors:  J J Hopwood; H Elliott
Journal:  Clin Sci (Lond)       Date:  1983-09       Impact factor: 6.124

7.  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

8.  A sensitive procedure for the diagnosis of N-acetyl-galactosamine-6-sulfate sulfatase deficiency in classical Morquio's disease.

Authors:  J Glössl; H Kresse
Journal:  Clin Chim Acta       Date:  1978-08-15       Impact factor: 3.786

9.  Biochemical defect of non-keratan-sulfate-excreting Morquio syndrome.

Authors:  A Fujimoto; A L Horwitz
Journal:  Am J Med Genet       Date:  1983-06

10.  Impaired degradation of keratan sulphate by Morquio A fibroblasts.

Authors:  J Glössl; H Kresse
Journal:  Biochem J       Date:  1982-04-01       Impact factor: 3.857

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  3 in total

1.  Glucuronate-2-sulphatase activity in cultured human skin fibroblast homogenates.

Authors:  C Freeman; J J Hopwood
Journal:  Biochem J       Date:  1991-10-15       Impact factor: 3.857

2.  A rare case of mucopolysaccharidosis.

Authors:  Ipsita Choudhury; Mona A Tilak; Arun Kumar Patra
Journal:  Indian J Clin Biochem       Date:  2013-06-08

3.  An At-Risk Population Screening Program for Mucopolysaccharidoses by Measuring Urinary Glycosaminoglycans in Taiwan.

Authors:  Hsiang-Yu Lin; Chung-Lin Lee; Yun-Ting Lo; Ru-Yi Tu; Ya-Hui Chang; Chia-Ying Chang; Pao Chin Chiu; Tung-Ming Chang; Wen-Hui Tsai; Dau-Ming Niu; Chih-Kuang Chuang; Shuan-Pei Lin
Journal:  Diagnostics (Basel)       Date:  2019-10-05
  3 in total

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