Association between clinical expression and molecular heterogeneity in β-thalassemia Tunisian patients

Mol Biol Rep. 2013 Nov;40(11):6205-12. doi: 10.1007/s11033-013-2732-y. Epub 2013 Sep 25.

Abstract

Beta-thalassemia is the most frequent hereditary blood disorder in Tunisia because of its geographic localization and history. This pathology is characterized by a complex multisystem process with genetic and biochemical interactions. The aim of this work was to establish phenotype/genotype association through studying the distribution and the relationship between β-thalassemia and α-thalassemia mutations and three polymorphic markers: the C → T polymorphism at -158 of the Gγ gene, the RFLP haplotype and the repeated sequence (AT)xTy in the β globin silencer, in two groups of β-thalassemia major and β-thalassemia intermedia (TI) patients. Statistical analysis has shown that moderate expression seen in TI patients was significantly associated to β(+) -87 (C → G), -30 (T → A) and IVSI-6 (T → C) mutations, haplotypes VIII, IX and Nb and to XmnI polymorphism. The regression analysis of combined genotypes (mutation/XmnI/RFLP haplotype) revealed that they contribute to justify 17.1 % of clinical expression diversity (p < 0.05). Among the studied genotypes the XmnI polymorphism seems to be the most determinant modulating factor, followed by the β-thalassemia mutation and RFLP haplotype. Our findings highlight the heterogeneity of molecular background of β-thalassemia that would be responsible of clinical variability.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • Gene Order
  • Genetic Association Studies*
  • Genetic Heterogeneity*
  • Haplotypes
  • Hemoglobins / genetics
  • Hemoglobins / metabolism
  • Humans
  • Middle Aged
  • Mutation
  • Nucleotide Motifs
  • Phenotype
  • Polymorphism, Genetic
  • Polymorphism, Restriction Fragment Length
  • Promoter Regions, Genetic
  • Tunisia
  • Young Adult
  • alpha-Thalassemia / genetics
  • alpha-Thalassemia / metabolism
  • beta-Globins / genetics*
  • beta-Thalassemia / blood
  • beta-Thalassemia / diagnosis*
  • beta-Thalassemia / genetics*
  • gamma-Globins / genetics

Substances

  • Hemoglobins
  • beta-Globins
  • gamma-Globins