Co-inheritance of α0 -thalassemia elevates Hb A2 level in homozygous Hb E: Diagnostic implications

Int J Lab Hematol. 2017 Oct;39(5):508-512. doi: 10.1111/ijlh.12677. Epub 2017 May 12.

Abstract

Introduction: Differentiation of homozygous hemoglobin (Hb) E with and without α0 -thalassemia is subtle on routine hematological ground. We examined in a large cohort of homozygous Hb E if the level of Hb A2 is helpful.

Methods: A total of 592 subjects with homozygous Hb E were recruited from ongoing thalassemia screening program. Additionally, five couples at risk of having fetuses with Hb Bart's hydrops fetalis who were homozygous Hb E were also investigated. Hb analysis was performed using capillary electrophoresis system. Globin genotypes were defined by DNA analysis.

Results: Subjects were classified into four groups including pure homozygous Hb E (n=532), homozygous Hb E/α0 -thalassemia (n=48), Hb Constant Spring EE Bart's disease (n=8), and Hb EE Bart's disease (n=4). The levels of Hb A2 were found, respectively, to be 4.97±0.69, 6.64±1.02, 4.86±0.87, and 7.60±1.04%. Among five couples at risk, α0 -thalassemia was identified in three subjects with Hb A2 >6.0%.

Conclusions: Increased Hb A2 level is a useful marker for differentiation of homozygous Hb E with and without α0 -thalassemia. This should lead to a significant reduction in number of referral cases of homozygous Hb E for molecular testing of α0 -thalassemia in routine practice.

Keywords: capillary electrophoresis; differential diagnosis; hemoglobin A2; hemoglobin E; α0-thalassemia.

MeSH terms

  • Adult
  • Biomarkers
  • Electrophoresis, Capillary
  • Erythrocyte Indices
  • Female
  • Genotype
  • Hemoglobin A2 / genetics*
  • Hemoglobin E / genetics*
  • Homozygote*
  • Humans
  • Hydrops Fetalis / diagnosis
  • Hydrops Fetalis / genetics
  • Inheritance Patterns*
  • Male
  • Mutation
  • alpha-Thalassemia / blood
  • alpha-Thalassemia / diagnosis*
  • alpha-Thalassemia / genetics*

Substances

  • Biomarkers
  • Hemoglobin A2
  • Hemoglobin E