Literature DB >> 6164767

Sanfilippo B syndrome (MPS III B): case report with analysis of CSF mucopolysaccharides and conjunctival biopsy.

A Federico, G Capece, A Cecio, N D'Auria, G Di Iorio, L Ronsisvalle, P Di Natale.   

Abstract

A case of a child with Sanfilippo B syndrome (MPS III B), born of a consanguineous marriage, is reported. Urinary mucopolysaccharide analysis showed an abnormal excretion mainly of heparan sulphate. N-acetyl-a-glucosaminidase activity was absent in the patient but was present in the heterozygous range in parents and siblings. CSF mucopolysaccharides were also abnormally high. In fibrocytes from conjunctival biopsy and CSF cells numerous vacuoles containing storage material were found. The presence of vacuoles in fibrocytes from conjunctival biopsy and/or in CSF cells can be useful in the diagnosis of many suspected lysosomal storage disorders.

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Year:  1981        PMID: 6164767     DOI: 10.1007/BF00313321

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  15 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.  [The results of the recent cytological studies of cerebrospinal fluid with sedimentation chamber].

Authors:  J SAYK
Journal:  Arztl Wochensch       Date:  1954-10-29

3.  Sanfilippo B syndrome (MPS III B): mild and severe forms within the same sibship.

Authors:  G Andria; P Di Natale; E Del Giudice; P Strisciuglio; P Murino
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4.  A newly recognized forme fruste of Hurler's disease (gargoylism).

Authors:  H G SCHEIE; G W HAMBRICK; L A BARNESS
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Review 5.  The systemic mucopolysaccharidoses.

Authors:  J Spranger
Journal:  Ergeb Inn Med Kinderheilkd       Date:  1972

6.  The systemic mucopolysaccharidoses. Ultrastructural and histochemical studies of conjunctiva and skin.

Authors:  K R Kenyon; H A Quigley; I E Hussels; R G Wyllie; M F Goldberg
Journal:  Am J Ophthalmol       Date:  1972-06       Impact factor: 5.258

7.  Sanfilippo B disease: serum assays for detection of homozygous and heterozygous individuals in three families.

Authors:  K von Figura; M Lögering; G Mersmann; H Kresse
Journal:  J Pediatr       Date:  1973-10       Impact factor: 4.406

8.  The sanfilippo B corrective factor: a N-acetyl-alpha-D-glucosamindiase.

Authors:  K von Figura; H Kresse
Journal:  Biochem Biophys Res Commun       Date:  1972-07-25       Impact factor: 3.575

9.  Clinical variability in Sanfilippo B disease: a report on six patients in two related sibships.

Authors:  J J van de Kamp; J F van Pelt; K O Liem; M A Giesberts; L T Niepoth; C R Staalman
Journal:  Clin Genet       Date:  1976-11       Impact factor: 4.438

10.  Mucolipidosis II (I-cell disease): ultrastructural observations of conjunctiva and skin.

Authors:  K R Kenyon; J A Sensenbrenner
Journal:  Invest Ophthalmol       Date:  1971-08
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6.  High-Throughput Liquid Chromatography-Tandem Mass Spectrometry Quantification of Glycosaminoglycans as Biomarkers of Mucopolysaccharidosis II.

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