Homozygous Hb Stanleyville-II [alpha2 78(EF7) Asn>Lys; HBA2:c.237C>A, not C>G] associated with genotype -α 3.7/-α 3.7 in two Brazilian families

Int J Lab Hematol. 2011 Dec;33(6):566-9. doi: 10.1111/j.1751-553X.2011.01321.x. Epub 2011 Apr 7.

Abstract

Introduction: Several hemoglobin variants have electrophoretic behavior similar to hemoglobin S, which may lead to false diagnosis for sickle-cell disorders in newborn screening programs. A homozygous hemoglobin with S mobility was detected in two unrelated babies in Brazil.

Methods: Isoelectric focusing and high-performance liquid chromatography assays, gene sequencing, and restriction fragment length polymorphism with AfeI were used to characterize the hemoglobin.

Results: Hb Stanleyville-II and -α(3.7) /-α(3.7) type I deletion in the α-globin gene was diagnosed. Parents were heterozygous for both Hb Stanleyville-II and α-thalassemia. Hypochromia and microcytosis were probably due to the homozygous α-thalassemia.

Conclusion: Stanleyville-II gene mutation is HBA2:c.237C>A, or C>G, and this information on the Globin Gene Server should be updated; AfeI test is a fast and accurate method to detect it; NBS programs should consider the possibility of Hb Stanleyville-II whenever IEF shows one band in the HbS position, and another one between S and C.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Base Sequence
  • Brazil
  • DNA Mutational Analysis
  • Family Health
  • Genotype
  • Hemoglobins, Abnormal / genetics*
  • Homozygote
  • Humans
  • Infant, Newborn
  • Molecular Sequence Data
  • Mutation, Missense*
  • alpha-Globins / genetics*
  • alpha-Thalassemia / genetics*

Substances

  • Hemoglobins, Abnormal
  • alpha-Globins
  • hemoglobin Stanleyville II