Literature DB >> 6435628

Isolation and characterization of N-acetylglucosamine 6-sulfate from the urine of a patient with Sanfilippo type D syndrome and its occurrence in normal urine.

J J Hopwood, H Elliott.   

Abstract

N-Acetylglucosamine 6-sulfate (GlcNAc6S) has been isolated from human urine and shown to be present at levels of approximately 0.02 and 14 mg/mmole creatinine in urine from normal individuals and a mucopolysaccharidosis type IIID (MPS IIID) patient respectively. We propose that the greater than 500-fold elevation of GlcNAc6S in urine from the MPS IIID patient indicates that this sulfated monosaccharide is also a substrate for the sulfatase deficient in MPS IIID patients. We further propose that part, if not all, of the GlcNAc6S found in urine may be produced from the cleavage by beta-N-acetylhexosaminidase A of non-reducing end beta-linked GlcNAc6S residues of keratan sulfate and/or sulfated glycoproteins.

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Year:  1983        PMID: 6435628

Source DB:  PubMed          Journal:  Biochem Int        ISSN: 0158-5231


  7 in total

1.  Impaired elastogenesis in Hurler disease: dermatan sulfate accumulation linked to deficiency in elastin-binding protein and elastic fiber assembly.

Authors:  A Hinek; S E Wilson
Journal:  Am J Pathol       Date:  2000-03       Impact factor: 4.307

2.  Caprine mucopolysaccharidosis IIID: a preliminary trial of enzyme replacement therapy.

Authors:  E Downs-Kelly; M Z Jones; J Alroy; K T Cavanagh; B King; R E Lucas; J C Baker; S A Kraemer; J J Hopwood
Journal:  J Mol Neurosci       Date:  2000-12       Impact factor: 3.444

Review 3.  Glycan-based biomarkers for mucopolysaccharidoses.

Authors:  Roger Lawrence; Jillian R Brown; Fred Lorey; Patricia I Dickson; Brett E Crawford; Jeffrey D Esko
Journal:  Mol Genet Metab       Date:  2013-07-29       Impact factor: 4.797

4.  Human liver N-acetylglucosamine-6-sulphate sulphatase. Catalytic properties.

Authors:  C Freeman; J J Hopwood
Journal:  Biochem J       Date:  1987-09-01       Impact factor: 3.857

5.  Human liver N-acetylglucosamine-6-sulphate sulphatase. Purification and characterization.

Authors:  C Freeman; P R Clements; J J Hopwood
Journal:  Biochem J       Date:  1987-09-01       Impact factor: 3.857

6.  N-acetylglucosamine 6-sulphatase deficiency in a Nubian goat: a model of Sanfilippo syndrome type D (mucopolysaccharidosis IIID).

Authors:  J N Thompson; M Z Jones; G Dawson; P S Huffman
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

7.  Urinary excretion of sulphated N-acetylhexosamines in patients with various mucopolysaccharidoses.

Authors:  J J Hopwood; H Elliott
Journal:  Biochem J       Date:  1985-08-01       Impact factor: 3.857

  7 in total

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