Literature DB >> 9435446

Liver microsomal transport of glucose-6-phosphate, glucose, and phosphate in type 1 glycogen storage disease.

P Marcolongo1, G Bánhegyi, A Benedetti, C J Hinds, A Burchell.   

Abstract

The transport of glucose-6-phosphate (G6P), glucose, and orthophosphate into liver microsomes, isolated from six patients with various subtypes of type 1 glycogen storage disease (GSD), was measured using a light-scattering method. We found that G6P, glucose, and phosphate could all cross the microsomal membrane, in four cases of type 1a GSD. In contrast, liver microsomal transport of G6P and phosphate was deficient in the GSD 1b and 1c patients, respectively. These results support the involvement of multiple proteins (and genes) in GSD type 1. The results obtained with the light-scattering method are in accordance with conventional kinetic analysis of the microsomal glucose-6-phosphatase system. Therefore, this technique could be used to directly diagnose type 1b and 1c GSD.

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Year:  1998        PMID: 9435446     DOI: 10.1210/jcem.83.1.4519

Source DB:  PubMed          Journal:  J Clin Endocrinol Metab        ISSN: 0021-972X            Impact factor:   5.958


  4 in total

1.  NPT4, a new microsomal phosphate transporter: mutation analysis in glycogen storage disease type Ic.

Authors:  D Melis; A C Havelaar; E Verbeek; G P A Smit; A Benedetti; G M S Mancini; F Verheijen
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

Review 2.  The glucose-6-phosphatase system.

Authors:  Emile van Schaftingen; Isabelle Gerin
Journal:  Biochem J       Date:  2002-03-15       Impact factor: 3.857

3.  A gene on chromosome 11q23 coding for a putative glucose- 6-phosphate translocase is mutated in glycogen-storage disease types Ib and Ic.

Authors:  M Veiga-da-Cunha; I Gerin; Y T Chen; T de Barsy; P de Lonlay; C Dionisi-Vici; C D Fenske; P J Lee; J V Leonard; I Maire; A McConkie-Rosell; S Schweitzer; M Vikkula; E Van Schaftingen
Journal:  Am J Hum Genet       Date:  1998-10       Impact factor: 11.025

4.  Histone 2A stimulates glucose-6-phosphatase activity by permeabilization of liver microsomes.

Authors:  Angelo Benedetti; Rosella Fulceri; Bernard B Allan; Pamela Houston; Andrey L Sukhodub; Paola Marcolongo; Brian Ethell; Brian Burchell; Ann Burchell
Journal:  Biochem J       Date:  2002-10-15       Impact factor: 3.857

  4 in total

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