Systemic gene dysregulation in classical Galactosaemia: Is there a central mechanism?

Mol Genet Metab. 2014 Nov;113(3):177-87. doi: 10.1016/j.ymgme.2014.08.004. Epub 2014 Aug 14.

Abstract

Classical Galactosaemia is a rare disorder of carbohydrate metabolism caused by a deficiency of galactose-1-phosphate uridyltransferase (GALT). The disease is life-threatening in the neonate, and the only treatment option is life-long dietary restriction of galactose. However, long-term complications persist in treated patients including cognitive impairments, speech and language abnormalities and premature ovarian insufficiency in females. Microarray analysis of T-lymphocytes from treated adult patients identified systemic dysregulation of numerous gene pathways, including the glycosylation, inflammatory and inositol pathways. Analysis of gene expression in patient-derived dermal fibroblasts of patients exposed to toxic levels of galactose, with immunostaining, has further identified the susceptibility of the glycosylation gene alpha-1,2-mannosyltransferase (ALG9) and the inflammatory gene annexin A1 (ANXA1) to increased galactose concentrations. These data suggest that Galactosaemia is a multi-system disorder affecting numerous signalling pathways.

Keywords: Biomarkers; Galactosaemia; Genomics; Glycosylation; Inflammation.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Annexin A1 / genetics
  • Annexin A1 / metabolism
  • Case-Control Studies
  • Cell Line
  • Female
  • Galactosemias / genetics*
  • Galactosemias / metabolism
  • Gene Regulatory Networks
  • Humans
  • Male
  • Mannosyltransferases / genetics
  • Mannosyltransferases / metabolism
  • Membrane Proteins / genetics
  • Membrane Proteins / metabolism
  • Oligonucleotide Array Sequence Analysis
  • T-Lymphocytes / metabolism
  • Transcriptome*
  • Young Adult

Substances

  • Annexin A1
  • Membrane Proteins
  • ALG9 protein, human
  • Mannosyltransferases