Literature DB >> 3161667

Debranching enzyme in fibroblasts, amniotic fluid cells and chorionic villi: pre- and postnatal diagnosis of glycogenosis type III.

O P van Diggelen, H C Janse, G P Smit.   

Abstract

Glycogenosis type III is characterized by a deficiency of debranching enzyme in most tissues, and it can be detected by the inability to liberate glucose from limit dextrin. However, using this assay, the deficiency is not expressed in cultured fibroblasts from patients with glycogenosis type III. We have demonstrated that the failure to detect debranching enzyme deficiency in fibroblasts is entirely due to interference of acid alpha-glucosidase, which can also hydrolyse limit dextrin. A method is described to remove specifically acid alpha-glucosidase allowing clear discrimination between fibroblasts from patients and controls, whereas heterozygotes showed intermediate values. The results with amniotic fluid cells and chorionic villi suggest the feasibility of first- and second-trimester prenatal diagnosis of glycogenosis III.

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Year:  1985        PMID: 3161667     DOI: 10.1016/0009-8981(85)90326-2

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


  4 in total

1.  Definitive prenatal diagnosis for type III glycogen storage disease.

Authors:  B Z Yang; J H Ding; B I Brown; Y T Chen
Journal:  Am J Hum Genet       Date:  1990-10       Impact factor: 11.025

2.  Polymorphic markers of the glycogen debranching enzyme gene allowing linkage analysis in families with glycogen storage disease type III.

Authors:  J Shen; H M Liu; Y Bao; Y T Chen
Journal:  J Med Genet       Date:  1997-01       Impact factor: 6.318

3.  First trimester prenatal diagnosis of glycogen storage disease type III.

Authors:  I Maire; G Mandon; M Mathieu
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

4.  A juvenile variant of glycogenosis IV (Andersen disease).

Authors:  A S Guerra; O P van Diggelen; F Carneiro; R M Tsou; S Simoes; N T Santos
Journal:  Eur J Pediatr       Date:  1986-08       Impact factor: 3.183

  4 in total

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