Literature DB >> 16125333

The molecular relationship between deficient UDP-galactose uridyl transferase (GALT) and ceramide galactosyltransferase (CGT) enzyme function: a possible cause for poor long-term prognosis in classic galactosemia.

Phiyani Justice Lebea1, Pieter J Pretorius.   

Abstract

Classic galactosemia is an autosomal recessive disorder that is caused by activity deficiency of the UDP-galactose uridyl transferase (GALT). The clinical spectrum of classic galactosemia differs according to the type and number of mutations in the GALT gene. Short-term clinical symptoms such as jaundice, hepatomegaly, splenomegaly and E. coli sepsis are typically associated with classic galactosemia. These symptoms are often severe but quickly ameliorate with dietary restriction of galactose. However, long-term symptoms such as mental retardation and primary ovarian failure do not resolve irrespective of dietary intervention or the period of initial dietary intervention. There seem to be an association between deficient galactosylation of cerebrosides and classic galactosemia. Galactocerebrosides and glucocerebrosides are the primary products of the enzyme UDP-galactose:cerebroside galactosyl transferase (CGT). There has been an observation of deficient galactosylation coupled with over glucosylation in the brain tissue specimens sampled from deceased classic galactosemia patients. The plausible mechanism with which the association between GALT and CGT had not been explained before. Yet, UDP-galactose serves as the product of GALT as well as a substrate for CGT. In classic galactosemia, there is a consistent deficiency in cerebroside galactosylation. We postulate that the molecular link between defective GALT enzyme, which result in classic galactosemia; and the cerebroside galactosyl transferase, which is responsible for galactosylation of cerebrosides is dependent on the cellular concentrations of UDP-galactose. We further hypothesize that a threshold concentration of UDP-galactose exist below which the integrity of cerebroside galactosylation suffers.

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Year:  2005        PMID: 16125333     DOI: 10.1016/j.mehy.2005.06.025

Source DB:  PubMed          Journal:  Med Hypotheses        ISSN: 0306-9877            Impact factor:   1.538


  9 in total

Review 1.  Classical galactosaemia revisited.

Authors:  Annet M Bosch
Journal:  J Inherit Metab Dis       Date:  2006-07-11       Impact factor: 4.982

2.  Biochemical changes and clinical outcomes in 34 patients with classic galactosemia.

Authors:  Tatiana Yuzyuk; Krista Viau; Ashley Andrews; Marzia Pasquali; Nicola Longo
Journal:  J Inherit Metab Dis       Date:  2018-01-19       Impact factor: 4.982

Review 3.  Remyelination in PNS and CNS: current and upcoming cellular and molecular strategies to treat disabling neuropathies.

Authors:  Sedigheh Momenzadeh; Mohammad-Saeid Jami
Journal:  Mol Biol Rep       Date:  2021-11-03       Impact factor: 2.316

4.  Classical Galactosaemia and CDG, the N-Glycosylation Interface. A Review.

Authors:  Ashwini Maratha; Hugh-Owen Colhoun; Ina Knerr; Karen P Coss; Peter Doran; Eileen P Treacy
Journal:  JIMD Rep       Date:  2016-08-09

5.  Oxidative stress contributes to outcome severity in a Drosophila melanogaster model of classic galactosemia.

Authors:  Patricia P Jumbo-Lucioni; Marquise L Hopson; Darwin Hang; Yongliang Liang; Dean P Jones; Judith L Fridovich-Keil
Journal:  Dis Model Mech       Date:  2012-07-05       Impact factor: 5.758

Review 6.  Sweet and sour: an update on classic galactosemia.

Authors:  Ana I Coelho; M Estela Rubio-Gozalbo; João B Vicente; Isabel Rivera
Journal:  J Inherit Metab Dis       Date:  2017-03-09       Impact factor: 4.982

7.  Deficits of facial emotion recognition and visual information processing in adult patients with classical galactosemia.

Authors:  Mirjam Korner; Sonja Kälin; Antoinette Zweifel-Zehnder; Niklaus Fankhauser; Jean-Marc Nuoffer; Matthias Gautschi
Journal:  Orphanet J Rare Dis       Date:  2019-02-26       Impact factor: 4.123

8.  Nucleotide sugar profiles throughout development in wildtype and galt knockout zebrafish.

Authors:  Minela Haskovic; Ana I Coelho; Martijn Lindhout; Fokje Zijlstra; Raisa Veizaj; Rein Vos; Jo M Vanoevelen; Jörgen Bierau; Dirk J Lefeber; M Estela Rubio-Gozalbo
Journal:  J Inherit Metab Dis       Date:  2020-06-05       Impact factor: 4.982

9.  From mind to mouth: event related potentials of sentence production in classic galactosemia.

Authors:  Inge Timmers; Bernadette M Jansma; M Estela Rubio-Gozalbo
Journal:  PLoS One       Date:  2012-12-26       Impact factor: 3.240

  9 in total

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