Literature DB >> 657539

Hypersialyloligosacchariduria in mucolipidoses: a method for diagnosis.

S Okada, T Kato, S Miura, H Yabuuchi, M Nishigaki, A Kobata, H Chiyo, J I Furuyama.   

Abstract

A method is described for the detection of abnormal oligosaccharides in a small (5 ml) volume of urine, employing filtration on a Bio Gel P-6 column, determination of neutral sugar and bound sialic acid, and determination of creatinine content. With this method increased urinary excretion of sialic acid-rich oligosaccharides has been detected in nine patients with mucolipidoses (five cases of mucolipidosis II and four patients of mucolipidosis, with beta-galactosidase deficiency). The filtration patterns of oligosaccharides in mucolipidoses were clearly distinguishable from those in other inborn errors of metabolism. Total excreted oligosaccharides were increased 5--30-fold in these patients; mucolipidosis II, 640--1350 microgram neutral sugar/mg creatinine; control 54 +/- 20 microgram neutral sugar/mg creatinine. The oligosaccharides consisted of three sialic acid-rich fractions and were common in both types of mucolipidosis. Our data indicate that hypersialyoligosacchariduria is the main biochemical feature of both types of mucolipidosis.

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Year:  1978        PMID: 657539     DOI: 10.1016/0009-8981(78)90129-8

Source DB:  PubMed          Journal:  Clin Chim Acta        ISSN: 0009-8981            Impact factor:   3.786


  9 in total

Review 1.  Sialidosis: a review of human neuraminidase deficiency.

Authors:  J A Lowden; J S O'Brien
Journal:  Am J Hum Genet       Date:  1979-01       Impact factor: 11.025

2.  Comprehensive urinary screening for inborn errors of complex carbohydrate metabolism.

Authors:  A C Sewell; J Gehler; J Spranger
Journal:  Klin Wochenschr       Date:  1979-06-01

3.  Infantile sialidosis: a phenocopy of type 1 GM1 gangliosidosis distinguished by genetic complementation and urinary oligosaccharides.

Authors:  R A Gravel; J A Lowden; J W Callahan; L S Wolfe; N M Ng Yin Kin
Journal:  Am J Hum Genet       Date:  1979-11       Impact factor: 11.025

4.  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

5.  Deficiency of neuraminidase in the sialidoses and the mucolipidoses.

Authors:  W R Den Tandt; J G Leroy
Journal:  Hum Genet       Date:  1980       Impact factor: 4.132

6.  A case of neuraminidase deficiency associated with a partial beta-galactosidase defect. Clinical, biochemical and radiological studies.

Authors:  S Okada; T Yutaka; T Kato; C Ikehara; H Yabuuchi; M Okawa; M Inui; H Chiyo
Journal:  Eur J Pediatr       Date:  1979-04-03       Impact factor: 3.183

7.  Human beta-galactosidase and alpha-neuraminidase deficient mucolipidosis: genetic complementation analysis of the neuraminidase deficiency.

Authors:  O T Mueller; T B Shows
Journal:  Hum Genet       Date:  1982       Impact factor: 4.132

8.  Urinary oligosaccharide excretion in disorders of glycolipid, glycoprotein and glycogen metabolism. A review of screening for differential diagnosis.

Authors:  A C Sewell
Journal:  Eur J Pediatr       Date:  1980-09       Impact factor: 3.183

9.  A mutation in a mild form of galactosialidosis impairs dimerization of the protective protein and renders it unstable.

Authors:  X Y Zhou; N J Galjart; R Willemsen; N Gillemans; H Galjaard; A d'Azzo
Journal:  EMBO J       Date:  1991-12       Impact factor: 11.598

  9 in total

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